chartered society of physiotherapy - association …...aims and scope: the apcp journal aims to...

Volume 9 Number 1 June 2018

ASSOCIATION OF PAEDIATRICCHARTERED PHYSIOTHERAPISTS

JOURNAL

Professional Network of the Chartered Society of Physiotherapy

EditorBarry Johnstone

Editorial Board:Assistant Editor VacantPrevious Editor Cate NaylorResearch Officer Sarah Westwater-WoodEducation Officer Anna Hebda-BoonNeonatal Fiona PriceNeuro-disability Claire TripathiNeuromuscular Marion MainMusculo-skeletal Luke WatsonRespiratory Esme TurnerCo-opted member Jo BrookOther Reviewers Sue Bush Catherine Duff Christina Calderon Claire Mayfield Dawn Pickering Jennifer McCahill Lesley Katchburian Pam Marmelstein Sue Allen Adare Brady Philip Harniess Sally Jary Marina DiMarco Naomi Winfield

Aims and scope: the APCP Journal aims to publish original research and other scholarly work related to paediatric physiotherapy – its scientific basis and clinical application, education of practitioners, management of services and policy.

For enquiries relating to the submission of articles contact:

APCPPO Box 99OkehamptonEX20 9AT

email: [email protected]: apcp.csp.org.uk

June 2018 APCP JOURNAL

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JOURNAL OF THE ASSOCIATION OF PAEDIATRIC CHARTERED PHYSIOTHERAPISTSVolume 9 Number 1 June 2018

CONTENTS

Editorial

Barry Johnstone – APCP Journal Editor ........................................................................................................................................... 2

Spinal Muscular Atrophy (SMA) type 1, a changing phenotype: Implications for motor function and physiotherapy management from the Nusinersen Expanded Access Program (EAP)

R. Tillmann, M. Main, L. Abbot, L. Edel, M. Scoto, F. Muntoni .................................................................................................... 4

Developmental outcome measures in neonatal physiotherapy services of the United Kingdom: a survey of current use, and facilitators and barriers to their implementation

B. Domislic, A. Harvey, H. Shannon ............................................................................................................................................... 13

Implementation and evaluation of an extended hours working pattern within an acute tertiary paediatric physiotherapy department

C. Ronan, D. Coggings, D. Carr ....................................................................................................................................................... 29

The Barriers and Facilitators to Implementing ‘The Daily Mile’™ in a UK Based Primary School: a Process Evaluation Protocol

J. Harris, L. Milnes, G. Mountain ..................................................................................................................................................... 37

Single case experimental design: maximising the potential for research within paediatric physiotherapy

G. Kelly, J. Shanley ............................................................................................................................................................................. 46

Development of a new preliminary item pool for children who are wheelchair users for the Pediatric Evaluation of Disability Inventory-Computer Adaptive Test (PEDI-CAT).

V. Knox ............................................................................................................................................................................................... 52

Rare Diseases – Jeune Syndrome

J. Thornton........................................................................................................................................................................................... 60

NICE Standard NG72

J. Kant, H. Cruickshank ..................................................................................................................................................................... 62

Submissions to the APCP Journal ................................................................................................................................................. 65

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Editorial – Exploring Research Priorities Barry Johnstone

Research is fundamental in supporting evidence based practice yet knowing where to start and what to research can be difficult. On the 1st March 2018, the CSP published their current research priorities for our profession. In order to develop the priorities physiotherapists, patients and carers were consulted. Previous priorities have been developed by the CSP in 1998, 2002 and most recently in 2012 (Rankin et al 2012). It is important that research priorities are updated to reflect current need to best deliver relevant evidence based practice for the profession. Changing demographics, changes to where care is delivered as well as advances in technology have all been highlighted by the CSP as to the need to continually review research priorities.

In the previous priority listing (Rankin et al 2012), priorities were categorised by specialty; musculoskeletal, neurology, cardiorespiratory and mental and physical health. Within the most recent list, these categories have been discontinued. The reason for this has not been elaborated on but perhaps suggests that many priorities can be shared within the profession. Furthermore, there appears to be a greater emphasis put on provision and access to services. Sixty-five priorities in total were developed (csp.org.uk/priorities) and the top 10 can be seen below;

1. When health problems are developing, at what point is physiotherapy most/least effective for improving patient results compared to no physiotherapy? What factors affect this?

2. When used by physiotherapists, what methods are effective in helping patients to make health changes, engage with treatment, check their progress, or manage their health after discharge?

3. What are the best ways to deliver physiotherapy services to meet patients’ needs and improve outcomes for patients and services?

4. To stop health problems occurring or worsening, what physiotherapy treatments, advice or approaches are safe and effective? Where more than one treatment/approach works, which work best and in what dose?

5. What are patients’ expectations regarding recovery, how do these compare to physiotherapists’ views and, where recovery is not possible, how is this managed?

6. How does waiting for physiotherapy affect patient and service outcomes?

7. What parts of physiotherapy treatments cause behaviour change or physical improvement?

8. What approaches are effective for enabling parents, relations or carers to support physiotherapy treatment or to help patients to manage their own health problem?

9. How is patient progress and/or the results of physiotherapy treatment measured? How is service performance measured and checked?

10. How can access to physiotherapy be improved for groups who have reduced access?

Although the priorities are not specific to paediatrics, most are applicable to the paediatric population and therefore can be a good starting point to developing a research idea. Furthermore, research ideas can be developed through governmental drivers including;

• Allied Health Professions into Action (NHS England 2017)

• Public Health Strategic Framework for Allied Health Professions (NHS Wales 2017)

• Ready to Act (Scottish Government 2016)

• A Strategy for Paediatric Healthcare Services Provided in Hospitals and in the Community (2016 – 2026) (Northern Ireland Department of Health 2016)

In this journal, we have 3 research papers on; outcomes following the administration of Nusinersen for children with spinal muscle atrophy (type 1), neonatal physiotherapists use of outcome measures and the development of the Pediatric Evaluation of Disability Inventory-Computer Adaptive Test for children who are wheelchair users. We also have a service review of of an extended hours working pattern within an acute tertiary paediatric

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physiotherapy department, a protocol for a process evaluation of a school-based activity programme and a review on single case experimental design within the context of paediatric physiotherapy.

Please do continue to send in papers for the journal, the deadline for the December 2018 journal is June 15th and if you would like to join our team of peer reviewers, please contact us via [email protected].

This is my first journal as editor and I would like to give a big thank you to Cate Naylor who has been of great support during my time as assistant editor and also for this edition of the journal. I am also grateful to the editorial board and the other reviewers.

References

NHS England, (2017). Allied Health Professions into Action.

NHS Wales, (2017). Public Health Strategic Framework for Allied Health Professions.

Northern Ireland Department of Health, (2016). A Strategy for Paediatric Healthcare Services Provided in Hospitals and in the Community (2016 – 2026).

Rankin, G., Rushton, A., Olver, P. and Moore, A., (2012). Chartered Society of Physiotherapy's identification of national research priorities for physiotherapy using a modified Delphi technique. Physiotherapy, 98(3), pp.260-272.

Scottish Government. (2016). Ready to Act A transformational plan for Children and young people, their parents, carers and families who require support from allied health professionals (AHPs).

Call for Abstracts!APCP Conference 2018 is in Manchester on 2-3 November 2018. Please submit abstracts for Free Papers/Posters by July 1st 2018.

Submission forms and advice are available from our website.

Please read the accompanying advice when writing your abstract and include all the information requested. Full details of methodology, data and analysis are needed for the committee to assess your submission. If sufficient information is not included,

your submission may be rejected. If you require advice to help you write your abstract prior to submission, please contact me.

Sarah Westwater-Wood, Research Officer

[email protected]

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Spinal Muscular Atrophy (SMA) type 1, a changing phenotype: Implications for motor function and physiotherapy management from the Nusinersen Expanded Access

Program (EAP)R. Tillmann [a, b, c *], M. Main [a, b, c], L. Abbot [b], L. Edel [b], M. Scoto [a, b, c], F. Muntoni [a, b, c]

[a] UCL Institute of Child Health, [b] Great Ormond Street Hospital, [c] NIHR Great Ormond Street Hospital Biomedical Research Centre.

*Corresponding author: [email protected]

ABSTRACT

Background

5q SMA, a neurodegenerative condition is caused by the loss of SMN1 gene resulting in reduced amount of SMN protein causing degeneration of motor neurons in the anterior horn cells. Nusinersen, an antisense oligonucleotide, is a new drug available in the UK on an expanded access program (EAP), to patients with SMA type 1. Nusinersen increases the production of SMN protein by the SMN2 gene, and has shown to increase motor function and survival in clinical trials. This study aims to assess changes in motor and respiratory function of the heterogeneous population on the EAP, and discuss resulting changes in management of SMA type 1 patients.

Method

Participants are patients treated at Great Ormond Street Hospital, registered under the SMA REACH UK study from March to October 2017. The CHOP-INTEND assessment was performed pre, post 4th and 5th injection, and information regarding respiratory support was taken from the medical notes.

Results

A total of 21 patients, aged 1 month to 9.5 years were taking part in the EAP. Statistically significant improvements in CHOP-INTEND scores were found for pre to post 5th injection for the total sample (p=0.001) and participants with 2 SMN2 copy numbers (p=0.004). No change was found for respiratory support needs.

Conclusion

Findings from our heterogeneous population show improvement in motor function, but no change was found for respiratory support needs as of yet. Nusinersen is not a cure, therefore a close relationship with the palliative team remains important. Additionally however, a proactive approach to contracture and scoliosis management, and good and safe nutrition to allow for overall well-being remain of paramount importance to maximally benefit from the treatment with Nusinersen. Therefore, standards of care will need to be reviewed to reflect this changing need.

Introduction

Spinal Muscular Atrophy (SMA) is an autosomal recessive neurodegenerative condition affecting 1 in 6000 to 1 in 11000 children per year (Finkel et al., 2017a, D'Amico et al., 2011). SMA is classified depending on the age of symptom onset as well as the highest motor function achieved (see table 1). While infants with SMA type 1 are unable to sit, children with SMA type 2 will have achieved independent sitting at some point but might lose this function over time, and children with SMA type 3 will be able to walk unaided but might lose this function throughout their youth while type 4 SMA patients are milder and diagnosed in adulthood (D'Amico et al., 2011).

As Nusinersen is currently only available to children with SMA type 1 in the UK, this paper will focus on

this group only. Children with SMA type 1 present with symptoms under 6 months of age, will never sit and have a life expectancy of under 2 years. Approximately 50-60 % are born with the most severe form labelled SMA type 1, and children will be very floppy and unable to lift their arms and legs, though some movement might be seen initially. Often only flickers of movement can be seen in the fingers, wrist or feet, if gravity is eliminated. Respiratory problems arise early with children presenting with abnormal patterns of breathing and requiring respiratory support from early on.

5q SMA is the most common genetic cause of childhood mortality. It is caused in 95% of cases by a homozygous deletion or mutation in the Survival Motor Neuron (SMN1) gene on chromosome 5q13 (Chiriboga et al., 2016, Finkel et al., 2017a). This loss

R. Tillmann / APCP Journal Volume 9 Number 1 (2018)

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of SMN1 results in decreased expression of Survival Motor Neuron protein (SMN) with consequent degeneration of Motor Neurons in the anterior horn cells and the brainstem causing loss of muscle function (Finkel et al., 2017a). Humans have a paralogous Survival Motor Neuron 2 (SMN2) gene also producing SMN protein. This gene however entails a C to T transition on exon 7, leading to 90-95% of transcribed mRNA to be truncated and the protein therefore being non-functional, see diagram 1 (Butchbach and

Burghes, 2004). As SMA patients do not have SMN1 gene they are left with the small amount of functional SMN protein produced by SMN2. This amount however is insufficient in compensating for the absent SMN1 production of SMN protein (Chiriboga et al., 2016). The amount of copy numbers of SMN2 largely defines severity of SMA symptoms, as a higher copy number indicates higher levels of SMN protein being available in the body (D'Amico et al., 2011).

Diagram 1- Difference between SMN1 and SMN2 genes in the general population and SMA patients. In the general population SMN1 will produce the majority of SMN protein. The main difference between SMN1 and SMN2 is a C to T conversion within exon 7, leading to reduced incorporation exon 7 in the SMN2 mRNA . This leads to incomplete protein (incomplete circles) and only a minority of functional SMN protein being produced.

SMA results from the loss of SMN1(cross) but retention of SMN2; SMN2, however, produces much less SMN protein than SMN1. Modified from (Butchbach and Burghes, 2004)

Table 1: SMA Classifications

SMA type Symptom Onset Max Motor Function SMN2 copy numbers

Life expectancy

0 Foetal Nil 1 Days - weeks

1 <6 m 1a: birth-2 weeks 1b: < 3m 1c: >3m

Never sit 1,2, 3 <2y

2 6-18 m Sit 2, 3, 4 10-40y

3 1.5y -18y 3a: < 3y 3b: >3y

Walk, but might lose ambulation

3, 4, 5 Normal

4 18y+ Normal function, some weakness

4,5 Normal

y= years, m= months, Max = maximum

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Nusinersen is an antisense oligonucleotide (Chiriboga et al., 2016, Chiriboga, 2017) designed to bind to the pre m-RNA of SMN2 enhancing the inclusion of exon 7 during its splicing, allowing SMN2 to produce more functional SMN protein (Chiriboga, 2017, Finkel et al., 2017a). Antisense oligonucleotide drugs cannot cross the blood brain barrier, and therefore Nusinersen needs to be administered via intrathecal injections (Chiriboga et al., 2016). Diagram 2 (copied from Chiriboga et al. (2016)) illustrates the function of an antisense oligonucleotide drug such as Nusinersen.

Findings from the ENDEAR study evaluating safety, feasibility and benefits of the drug favoured the intervention group as 41% of participants showed motor milestone responses versus 0% in the control group, in the interim analysis. This was conducted when approximately 80 participants were enrolled in the study for at least 6 months (Finkel et al., 2017a). Due to these findings the placebo arm of the study was discontinued and all placebo patients were enrolled into an open label study receiving the active compound (Finkel et al., 2017a). The drug, commercial name Spinraza, has received Food and Drug Administration (FDA, USA) and European Medicines Agency (EMA) approval.

In the final analysis of this study, the response rate in motor milestone achievement increased even more, to 53% for the intervention versus 0% in the control

group. Additionally, the intervention group was favourable for event free survival for either death or permanent ventilation, as well as overall survival at 0.53, p=0.005 and 0.37, p=0.004 respectively (Finkel et al., 2017a).

In another study patients with SMA type 2 and 3 receiving Nusinersen showed significant changes on the Hammersmith Functional Motor Scale Extended (HFMSE) (Chiriboga et al., 2016). While the natural history data suggest a decline of -1 point on the HFMSE over 2 years, participants in this study had improved their HFMSE score at 9-14 month follow-up by a mean change of +5.8 points (Mercuri et al., 2016, Chiriboga, 2017).

Overall improvements in motor function were seen for all SMA type 1,2 and 3 patients, with best outcomes observed for pre-symptomatic treatment (Chiriboga, 2017).

Studies enrolling SMA type 1 patients used the Children`s Hospital of Philadelphia – Infant test of Neuromuscular Conditions (CHOP-INTEND) to establish change in motor function. This test assesses gross motor function in the very weak child with SMA type 1 and has been validated for use in this population (Glanzman et al., 2010, Glanzman et al., 2011). Findings from the NURTURE study, which enrolled pre-symptomatic infants with genetically

Diagram 2- Copied from (Chiriboga et al., 2016). Antisense Oligonucleotide Intron connects to C to T transition in the SMN2 gene allowing inclusion of exon 7 in the mRNA and therewith increased production of full length SMN protein.

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confirmed SMA having 2 or 3 SMN2 copy numbers, equally showed improvements in motor function as measured with the CHOP INTEND as well as improved respiratory function (Hwu et al., 2017).

Currently in the UK children with a genetically and clinical confirmed diagnosis of SMA type 1 can receive Nusinersen under the EAP, where local trust approval was gained. At Great Ormond Street Hospital (GOSH), the EAP has been running since March 2017. While the inclusion criteria for the studies were very strict, the EAP allows administration of Nusinersen to SMA type 1 patients irrespective of their motor function, respiratory status or age. At the Great Ormond Street Institute of Child Health (GOS ICH) we currently run an ongoing natural History Study for children with SMA, called SMA REACH (Research And Clinical Hub, http://www.smareachuk.org/). All children with a diagnosis of SMA, with Types 1 to 3 are eligible. It is a database study, by which data is collected for SMA Type 2 and 3 using the Revised Hammersmith Scale (Ramsey et al., 2017), and the Revised Upper Limb Module (Mazzone et al., 2017). Both of these tests have shown to be reliable tools with good psychometric properties (Mazzone et al.,

2017, Ramsey et al., 2017). For children with SMA type 1 the CHOP Intend as well as range of motion are collected together with other relevant medical information (Glanzman et al., 2010, Glanzman et al., 2011). Currently the anonymized data is stored on UCL computers. However future plans are to enable uploading of the data to a national database that can be used to answer specific research and clinical questions.

The aims of this study were therefore to present findings from a heterogeneous sample in regards to changes in motor and respiratory function, and describe the implications for management of children with SMA type 1 for paediatric physiotherapists.

Methods

This project was conducted under the SMA REACH UK study, for which ethical approval was obtained. SMA REACH UK is a natural history study for all SMA types. The inclusion criteria for this study were the same as the eligibility criteria for the EAP. The EAP allows access to Nusinersen for patients with a genetically confirmed homozygous deletion or non-

Table 2: Dosing Schedule Nusinersen

Age Dose Volume

0-3 9.6mg 4ml

3-6m 10.3mg 4.29ml

6-12m 10.8mg 4.5ml

12-24m 11.3mg 4.71ml

Older than 24m 12mg 5ml

mg- milligram, ml- millilitre, m- months

Table 3: Participant Demographics

Participants (N)

SMA type 1 21

SMN2 number23NK

1452

GenderMF

911

Age Median(range) 16m (1m -9.5y)

Number of completed injections1345

11108

(N)- number of participants, NK- not known, M- male, F- female, m- months, y- years

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function mutation of SMN1 and a clinical diagnosis compatible with SMA type 1. It has been running at GOSH since March 2017, and children enrolled between March and October 2017 were included in this study.

Drug administration occurred on day 1, 15, 30, and 60 for the loading phase, followed by repeat intrathecal injections every 4 months. The dosing regimen can be found in table 2. The drug administration schedule and dosing have remained the same as in the original ENDEAR (now open label SHINE) study (Finkel et al., 2017).

The CHOP-INTEND was developed by Glanzman et al. (2010) for the assessment of motor function in infants with severe neuromuscular conditions, the full assessment is freely available from the appendix of bespoke article(Glanzman et al., 2010). It has good inter-rater reliability (ICC= 0.98) for children with SMA Type 1, and normally developing infants (ICC=0.93) (Glanzman et al., 2010). Intra-rater reliability was equally high (ICC= 0.96 and) (Glanzman et al., 2011). In the validation of the scale the CHOP-INTEND was negatively correlated to SMN2 copy number, indicating lower CHOP-INTEND scores with less SMN2 copies, age, and respiratory support (r=-0.60, r=-0.51, r=-0.74) respectively (Glanzman et al., 2011). Even though the CHOP-INTEND was initially intended for infants, it is currently also used on older children with SMA type 1 receiving Nusinersen, as it is the only assessment available to assess motor function over time for very weak SMA type 1 patients (Chiriboga, 2017). The CHOP-INTEND was also used in the clinical trials evaluating the effect of Nusinersen and is recommended to be used for children entering the EAP to assess change in motor function. The CHOP-INTEND Protocol was followed (Glanzman et al., 2010) . The assessment was conducted before the first injection, after the 4th injection and then

systematically after each following injection. Three Clinical and one research Physiotherapist were trained to conduct the CHOP-INTEND assessment. The Clinical Physiotherapists were trained clinically by the Lead Physiotherapist and the research physiotherapist were trained for reliability of CHOP-INTEND performance as part of Clinical trials in SMA. Assessments were conducted by the same assessor, unless that person was unavailable for any reason (i.e. absence, sickness annual leave) in which case one of the other Physiotherapists would take over. As children under the EAP are seen clinically it was felt that the clinical physiotherapists were the main professionals to be involved in the assessments.

Medical notes of each enrolled child were viewed for information regarding the respiratory status pre, post 4th and 5th dose. Respiratory status, was recorded descriptively as presented in the medical notes. The information from the closest assessment to the injection was taken.

SPSS 24 was used for statistical analysis. Paired sample t-test was conducted for pre- to post 4th injection and pre- to post 5th injection. Paired sample t-test was conducted for pre- to post 4th and post 5th injection by SMN2 copy number. This was done, as patients had either pre and post 4th or pre and post 5th injection CHOP-INTEND scores, and therefore ANOVA could not be used.

Results

A total of 21 patients were registered on the EAP (from March to October 2017), see table 3 for demographic information. Data was included for all injections prior to the 1.11.2017. The loading phase was completed by 10 participants. Two children were in their loading phase with 1 and 3 injections and therefore did not have post 4th injection assessments. One child could

Table 4: Results CHOP-INTEND pre and post 4th and 5th injection

Pre CHOP Mean (SD)

Post-4th CHOP Mean (SD)

p-value (95%Ca) Post-5th CHOP Mean (SD)

p-value (95%Ca)

Descriptive total sample

N:2026.25 (10.41)

N:833.38 (10.02)

N:641.17 (6.14)

Paired sample t-test

N:828.25 (10.93)

N:833.38 (10.02)

p= 0.061 95%CI (-10.56-0.319)

N:542.80 (5.21)

p=0.001 95%CI(-8.75-(-9.83))

SMN2 copy number2copies

N:425.50 (7.50)

N:4 33.25(8.05)

p= 0.096 95%CI(-18.011-2.51)

N:440.75(2.87)

p= 0.004 95%CI(-16.50-(-7.87)NA

3copies N:337.67 (6.11)

N:339.00 (8.54)

p=0.776 95%CI(-18.95-16.29)

N:151

N=number of participants, SD=Standard deviation, CHOP= CHOP-INTEND, 95%CI= 95%Confidence Interval, p= statistically significant at 0.05, NA=Not applicable

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Graph 2: CHOP-INTEND scores by participant for pre 1st injection and post 5th injection comparison. Line at 40 expected upper limit for SMA type 1

Graph 1: CHOP-INTEND scores by participant for pre 1st injection and post 4th injection comparison. *Triangle same value pre and post 4th dose. Line at 40 expected upper limit for SMA type 1

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not be tested prior to the first injection due to excessive crying and lack of cooperation, but had a post 4th injection assessment with the CHOP-INTEND. See graph 1 and 2 for change in CHOP-INTEND scores by participant.

Mean and Standard deviation (SD) for all participants (N) for CHOP-INTEND Scores were, 26.25 (SD:10.41, N:20) for pre-injection, 33.38 (10.02, N:8) post 4th injection, and 41.17(6.14, N:6) post 5th injection.

The change in CHOP INTEND Score approached statistically significance for pre 1st injection to post 4th injection (N:8, p=0.061 95%CI[-10.56-0.319]), and was statistically significantly different for pre 1st injection to post 5th injection (N:5, p=0.001, 95%CI[-8.75- (-9.83)]). If split by SMN2 copy number, again statistical significance was reached from pre 1st injection to post 5th Injection for 2 SMN2 copy numbers (N:4, p= 0.004 95%CI[-16.50-(-7.87)]), see table 4.

Eight out of 20 children did not need any respiratory support prior to the start of Nusinersen, 6/8 of whom

were still not using respiratory support post 4th dose. For 1 child no post 4th dose clinic letter was available stating respiratory support needs, and one child had an acute chest infection and it was unclear at this point in time if the ventilator support of 14h was due to disease progression or secondary to the respiratory event, see table 5 for respiratory status.

Discussion and Implications for Physiotherapy management

Our study of a heterogeneous sample with SMA type 1, showed statistically significant improvements on the CHOP-INTEND from 1st to 5th dose, similar to results presented in the literature, even though children with chronic symptoms such as contractures, scoliosis, and of older age, unlike participants in the clinical trials were included (Chiriboga, 2017, Chiriboga et al., 2016, Finkel et al., 2017a). Considering that approximately 50-60% of children born with SMA are diagnosed with

the severe type 1, their presentation and therewith their treatment will need to adapt to the changing needs. In addition to palliative care, proactive and preventative treatment are becoming important, especially for contracture and scoliosis prevention and management. When planning spinal surgery, it can be an option to discuss sparing of specific lumbar vertebrae to ensure intrathecal injections can be given in the future.

Even though the improvements seen in the treated children are exciting and indeed very unexpected for children with SMA type 1, this can cause unforeseen difficulties. Even with gaining functional abilities of the arms and legs some children in our sample did not gain head control as of yet. These children are getting older and stronger, and equipment such as car seats addressing the needs of the child, such as being transported in the reclined or lying position can be more and more difficult to find. Especially if respiratory support is required, adequate equipment might be hard to find.

Overall our sample did not show improvements in

respiratory status. One child started the program without any respiratory support but was on 14 hours at the post 4th injection assessment, it could not be established if this was due to disease progression or an acute respiratory infection. It is still unknown which effect Nusinersen will have on respiratory function in the long term in children who lived with the symptoms for longer, and if lost respiratory function can be regained later on in life. Findings from a study in pre-symptomatic infants, presented at the recent WMS suggested that early treatment is most beneficial, and indeed showed favourable outcomes for respiratory function in comparison to the natural history (Hwu et al., 2017). This raises the question for the need of new-born screening, which is currently not available in the UK.

In the UK, only children diagnosed postnatal can currently receive Nusinersen, one child in our sample received their first injection at 1 month, which is


Table 5: Descriptive information for ventilatory support

pre Post loading dose Change

Ventilatory dependent 6 6 NA

Ventilated 14h 1 2 1 changed from no ventilation to 14h (due to respiratory infection)

Nocturnal 5 5 NA

No ventilatory support 8 6 1 changed to 14h due to respiratory infection

NA= not applicable, no separation between post 4th and 5th injection as information was taken from last available clinic letter stating respiratory status, therefore not exact for either 4th or 5th injection

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felt to be favourable in response to the drug and improvements in motor development. However, several patients currently treated have lived with their symptoms for many month or years and it remains questionable if a long-term benefit for those patients in regards to either respiratory or motor function can be achieved. Currently we do not know how contractures or compromised respiratory function will affect the overall benefits of the drug to the patient, or if lost skills and function can be regained when being on the drug. It therefore remains questionable if treatment of patients with chronic symptoms will be favourable in a cost versus benefit analysis, especially as long-term risks, and complications are not established at present.

The effects of Nusinersen on motor, respiratory function and quality of life for treated children will need careful evaluation over time. This requires physiotherapists and clinicians working with these children to ensure adequate documentation of progress and presentation is conducted for both children on the drug as well as children whose families have opted not to receive Nusinersen.

Additionally, adequate assessment and documentation is highly important in light of the possible approval for Nusinersen by the NHS England, as this drug has a very high cost per child per year, it is highly likely that data will need to be presented at a later stage to prove the benefits of the drug in the long term. In fact, the EMA press release (April 2017) states that post marketing data will be collected to allow ongoing evaluation of long-term safety, feasibility and benefit of the drug.

The drug has passed safety and tolerability studies and is indeed available commercially in the US and several EU countries, however long-term data is not yet available due to the relatively short span since Nusinersen was initially tested. We therefore do not know if repeat intrathecal injections will pose a danger, or if the beneficial effect will continue to slow down disease progression in the long term (King and Bishop, 2017). As other European countries allow SMA type 1,2 and 3 patients in the EAP, future studies should address feasibility, safety and benefit of long-term treatment with Nusinersen.

The results as presented in the literature and our findings are promising, indicating the need to implement reviewed standards of care for infants with SMA type 1, to address the additional needs.

The Neuromuscular Committee of the APCP and the ENMC have published guidance and advice recently (Committee, 2017, Finkel et al., 2017b). Palliative care will remain an important part of the care for children with SMA type 1, but proactive and preventative interventions like contracture prevention,

management of scoliosis, optimal nutrition, optimal respiratory status, maintenance of good lung health and overall well-being, are necessary to ensure the best possible outcome can be achieved when starting Nusinersen.

Limitations of the study

Due to the small sample size and no overlap between participants having an assessment post 4th and post 5th injection ANOVA could not be used for statistical analysis and instead two separate t-test had to be performed, bootstrapping was applied to account for multiple comparisons.

Conclusion

In conclusion, the best outcome is to be expected from pre-symptomatic treatment, however even our heterogeneous population with children of different ages and with different starting points showed improvements in their motor function on the CHOP-INTEND.

At present we do not know the long-term outcomes, possible complications or how Nusinersen will affect the quality of life of children with SMA type 1 and their families. Therefore, careful and accurate documentation, especially under the light of possibly needing to prove the benefits of the drug, in a re-evaluation likely to happen, should NHS England agree the funding of Nusinersen, are an important aspect when being involved in the care of children with SMA type 1.

For Physiotherapists and Clinicians working with children with SMA this implies a new set of standards of care with early contracture and scoliosis preventative interventions as described by the APCP Neuromuscular Committee and the ENMC (Commitee, 2017, Finkel et al., 2017b). It also raises the question for benefit of rehabilitative and developmental interventions in optimising the child`s potential benefit of Nusinersen.

While this is a very exciting time for SMA, as Nusinersen is changing the phenotypes as previously known, no long-term data of the benefits of Nusinersen is currently available. Good documentation of assessments to establish, longitudinal change, potential harm, patient populations with the greatest benefit, will help to further evaluate the long-term benefits of the drug and hopefully answer some of the questions we currently have no answers to.

Acknowledgments

This research was supported by the NIHR Great Ormond Street Hospital Biomedical Research Centre. The views expressed are those of the author(s) and not necessarily those of the NHS, the NIHR or the Department of Health.


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BUTCHBACH, M. E. R. & BURGHES, A. H. M. 2004. Perspectives on models of spinal muscular atrophy for drug discovery. Drug Discovery Today: Disease Models, 1, 151-156.

CHIRIBOGA, C. A. 2017. Nusinersen for the treatment of spinal muscular atrophy. Expert Rev Neurother, 17, 955-962.

CHIRIBOGA, C. A., SWOBODA, K. J., DARRAS, B. T., IANNACCONE, S. T., MONTES, J., DE VIVO, D. C., NORRIS, D. A., BENNETT, C. F. & BISHOP, K. M. 2016. Results from a phase 1 study of nusinersen (ISIS-SMN(Rx)) in children with spinal muscular atrophy. Neurology, 86, 890-7.

COMMITEE, N. 2017. Guidance for paediatric physiotherapists managing Neuromuscular disorders. APCP.

D'AMICO, A., MERCURI, E., TIZIANO, F. D. & BERTINI, E. 2011. Spinal muscular atrophy. Orphanet J Rare Dis, 6, 71.

FINKEL, R. S., MERCURI, E., DARRAS, B. T., CONNOLLY, A. M., KUNTZ, N. L., KIRSCHNER, J., CHIRIBOGA, C. A., SAITO, K., SERVAIS, L., TIZZANO, E., TOPALOGLU, H., TULINIUS, M., MONTES, J., GLANZMAN, A. M., BISHOP, K., ZHONG, Z. J., GHEUENS, S., BENNETT, C. F., SCHNEIDER, E., FARWELL, W. & DE VIVO, D. C. 2017a. Nusinersen versus Sham Control in Infantile-Onset Spinal Muscular Atrophy. N Engl J Med, 377, 1723-1732.

FINKEL, R. S., SEJERSEN, T. & MERCURI, E. 2017b. 218th ENMC International Workshop: Revisiting the consensus on standards of care in SMA. Neuromuscular Disorders, 27, 596-605.

GLANZMAN, A., MAZZONE, E., MAIN, M., PELLICCIONI, M., WOOD, J., SWOBODA, K., SCOTT, C., PANE, M., MESSINA, S. & BERTINI, E. 2010. The children’s hospital of philadelphia infant test of neuromuscular disorders (CHOP INTEND): test development and reliability. Neuromuscular Disorders, 20, 155-161.

GLANZMAN, A. M., MCDERMOTT, M. P., MONTES, J., MARTENS, W. B., FLICKINGER, J., RILEY, S., QUIGLEY, J., DUNAWAY, S., O'HAGEN, J. & DENG, L. 2011. Validation of the Children's Hospital of Philadelphia Infant Test of Neuromuscular Disorders (CHOP INTEND). Pediatric Physical Therapy, 23, 322-326.

HWU, W., DE, D., BERTINI, E., FOSTER, R., GHEUENS, S., FARWELL, W. & REYNA, S. 2017. P.384 - Outcomes after 1-year in presymptomatic infants with genetically diagnosed spinal muscular

atrophy (SMA) treated with nusinersen: interim results from the NURTURE study. Neuromuscular Disorders, 27, S212.

KING, N. & BISHOP, C. 2017. New treatments for serious conditions: ethical implications. Gene Therapy.

MAZZONE, E. S., MAYHEW, A., MONTES, J., RAMSEY, D., FANELLI, L., YOUNG, S. D., SALAZAR, R., DE SANCTIS, R., PASTERNAK, A., GLANZMAN, A., CORATTI, G., CIVITELLO, M., FORCINA, N., GEE, R., DUONG, T., PANE, M., SCOTO, M., PERA, M. C., MESSINA, S., TENNEKOON, G., DAY, J. W., DARRAS, B. T., DE VIVO, D. C., FINKEL, R., MUNTONI, F. & MERCURI, E. 2017. Revised upper limb module for spinal muscular atrophy: Development of a new module. Muscle Nerve, 55, 869-874.

MERCURI, E., FINKEL, R., MONTES, J., MAZZONE, E. S., SORMANI, M. P., MAIN, M., RAMSEY, D., MAYHEW, A., GLANZMAN, A. M., DUNAWAY, S., SALAZAR, R., PASTERNAK, A., QUIGLEY, J., PANE, M., PERA, M. C., SCOTO, M., MESSINA, S., SFRAMELI, M., VITA, G. L., D'AMICO, A., VAN DEN HAUWE, M., SIVO, S., GOEMANS, N., KAUFMANN, P., DARRAS, B. T., BERTINI, E., MUNTONI, F. & DE VIVO, D. C. 2016. Patterns of disease progression in type 2 and 3 SMA: Implications for clinical trials. Neuromuscul Disord, 26, 126-31.

RAMSEY, D., SCOTO, M., MAYHEW, A., MAIN, M., MAZZONE, E. S., MONTES, J., DE SANCTIS, R., DUNAWAY YOUNG, S., SALAZAR, R., GLANZMAN, A. M., PASTERNAK, A., QUIGLEY, J., MIREK, E., DUONG, T., GEE, R., CIVITELLO, M., TENNEKOON, G., PANE, M., PERA, M. C., BUSHBY, K., DAY, J., DARRAS, B. T., DE VIVO, D., FINKEL, R., MERCURI, E. & MUNTONI, F. 2017. Revised Hammersmith Scale for spinal muscular atrophy: A SMA specific clinical outcome assessment tool. PLoS One, 12, e0172346.

EMA press release, accessed 28.12.2017: http://www.ema.europa.eu/docs/en_GB/document_library/Press_release/2017/04/WC500226270.pdf

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Bozana Domislic [a], Alex Harvey [a, b], Harriet Shannon [a *][a] UCL Great Ormond Street Institute of Child Health.

[b] Department of Clinical Sciences, Brunel University London.*Corresponding author: [email protected]

ABSTRACT

Background and Purpose

Current guidelines recognise the need for neonatal physiotherapists to provide surveillance assessments to neonates, from birth onwards. This study explored the current use, barriers and facilitators to using developmental outcomes measures in neonatal units, in the United Kingdom.

Method

A cross-sectional, web-based survey with 32 items was piloted and distributed to members of the Association of Paediatric Chartered Physiotherapists, and advertised on social media and professional discussion forums. Responses from the group of therapists who reported using outcome measures, were compared with those who reported not using them, using Mann-Whitney U tests for non-parametric data. Other findings were presented descriptively.

Results

Forty-three completed surveys were analysed. Most respondents (91%) had a positive attitude towards the use of developmental outcome measures, and many (79%) used them in their neonatal practice. These included the General Movement Assessment (n=18/34), Hammersmith Neonatal Neurological Examination (n=16/34), and Lacey Assessment of Preterm Infants (n=15/34). The high cost of acquiring certain measures was a perceived barrier (86%). The presence of a neonatal-specialist physiotherapist (p=0.023), active engagement in continuous professional development (0.011) and support from fellow physiotherapists (p<0.001) significantly influenced outcome measure utilisation.

Conclusion

Outcome measures were commonly used in the analysed units. Perceived utility and positive attitudes towards outcome measure use are in-line with current recommendations. This must be weighed up against the high acquisition cost of some measures. Further research is required to define tailored strategies for promoting best practice in the utilisation of specific developmental outcomes in neonatal care.

Introduction

Approximately 60,000 babies are born prematurely (earlier than 37 weeks gestation), in the United Kingdom (UK) every year (Office of National Statistics, 2016). There has been continued improvement in the delivery of neonatal care that has enabled an increase in survival of infants born extremely prematurely between both 1995 and 2006, and still further between 2008 and 2014 (Santhakumaran et al, 2017). However, infants who survive a premature birth are at greater risk of developmental delay with associated long-term consequences, such as cerebral palsy (Moore et al, 2012). Physiotherapists play a vital role in the treatment of neonates in the UK, particularly those who are at increased risk of developmental delay. Input from a physiotherapist may include baseline assessment, parental education and working within the multi-

disciplinary team to ensure that the complex needs of the family are being met. Guidelines published by the National Institute for Health and Care Excellence (NICE) on the developmental follow-up of children and young people born prematurely, recommended that premature babies are monitored for the first two years of their lives (NICE, 2017). Physiotherapists are well placed to contribute to this monitoring process.

The physiotherapy assessment of neonates can provide an important baseline of information. This can then be used to monitor developmental evolution, identify abnormalities and predict developmental disabilities (Majnemer and Mazer, 1998). Therefore, the use of a valid, reliable, standardised tool has the potential to highlight those patients requiring early intervention, aid in decision-making processes regarding the management of the baby, and ensure

H. Shannon / APCP Journal Volume 9 Number 1 (2018)

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optimal quality of care (CSP, 2012). However, the purpose of outcome measures in neonates varies widely, encompassing neurological, neurobehavioural and motor performance assessments. They also have different functions. These include predictive value (the ability to predict the likely future course of development), evaluation (change as a result of a specific intervention) and a discriminative function (the ability to discriminate between the presence or absence of a certain impairment at a single time-point). For these reasons, the utilisation of developmental outcome measures is likely to vary substantially between hospitals. This could be problematic when the infant is transferred between neonatal units, as 10% of hospitalised infants were in 2015 (RCPCH, 2016).

To date, no studies have investigated the current use of, or facilitators and barriers to, using outcome measures in neonatal care. A previous cross-sectional survey found that 89.1% from a sample of 97 physiotherapists used a standardised assessment tool during their neurodevelopmental follow-up programmes of neonates (Harniess and Nikopoulou-Smyrni, 2015). The inclusion criteria for this particular work specified that ‘lead’ neonatal physiotherapists would be recruited, therefore it remains unknown whether ‘non-lead’ physiotherapists have a similar approach to the use of outcome measures. Furthermore, the study concentrated on follow-up assessment, leaving the broader use of outcome measures unknown.

Although barriers to using outcome measures have been identified in some specialties of physiotherapy (Swinkels et al, 2011), the unique environment of neonatal care means that some of the challenges are likely to be specific to the assessment of neonates. It is vital that the use of outcome measures in the UK is quantified, to measure the extent to which best practice is currently being adhered to. Barriers should be identified so that strategies can be implemented to address them. ‘Facilitators’ (factors that enable or support therapists to utilise outcome measures), will provide researchers and clinicians with an understanding of how best to encourage others to utilise outcomes. Therefore, the aims of the study were threefold. Firstly, to capture a snapshot of current use of developmental outcome measures in neonatal care in the UK. Secondly, to identify any facilitators or barriers that supported or impeded their implementation. Thirdly, to describe differences in neonatal unit, or in attitudes and approaches between those therapists who used outcome measures, and those who did not.

Method

Ethical approval was granted by the Chair of the UCL Research Ethics Committee on 6th June 2017

(REC reference 11151/001). UCL Data Protection and Risk Assessment registration was obtained on 26th May 2017 (reference no. Z6364106/2017/05/124). To prevent the occurrence of duplicate responses from the same hospital, participants were requested to provide demographic details of their units, which were coded to maintain anonymity in accordance with the Data Protection Act 1998.

A survey was selected as the most appropriate method for meeting the aims of the study. It comprised of 32 items divided into three domains: the use of developmental outcome measures; barriers and facilitators to their implementation; neonatal physiotherapy service provision and training (Appendix 1). Inclusion criteria for completing the survey were physiotherapists working in neonatal care in the UK, with only one response permitted per hospital site.

The survey included closed-ended multiple-choice questions, with additional free-text space for respondents to clarify their responses or add any options that were not listed (Polgar and Thomas, 2013). Perceived facilitators and barriers to implementation of outcome measures were gathered using a 5-point Likert scale, with responses ranging from ‘strongly disagree’ (1 point) to ‘strongly agree’ (5 points) in response to a range of statements. These statements were based on previous studies into the implementation of guidelines and outcome measures for general practitioners, midwives and physiotherapists (Peters et al., 2003, Van der Wees et al., 2013, Van Peppen et al., 2008). The final section of the survey was adapted from a validated questionnaire published in 2012, the aim of which was to establish a benchmark for the overall provision of neonatal physiotherapy services in the UK (Ronan and Barron, 2012). With permission from the principal author, eight questions from that questionnaire were utilised in the current study, in order to gather data regarding neonatal units, levels of expertise and training.

The questionnaire was piloted among two specialist neonatal physiotherapists who were independent of the study, and three members of UCL academic staff. This helped to identify ambiguous questions, and any perceived biased or leading questions. The participants of the pilot study were also asked to email their comments regarding the time taken to complete the survey and the clarity, relevance and completeness of the survey in order to evaluate its face and content validity (Polgar and Thomas, 2013). Three items were added (items 14, 18 and 19, see Appendix 1) and details within the survey, in particular the order of the domains and the phrasing of the questions were modified. One specific point raised by all reviewers related to the frequent use of ‘negative sentences’ such as, ‘the use of the developmental outcome measures

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is not too time-consuming.’ Although reverse worded items have been shown to reduce response bias, recent evidence suggests that they are more likely to result in confusion (Sonderen et al, 2013). The reviewers also had differing opinions over how to respond to such questions. Therefore, for clarity, these were re-phrased to provide consistently positively-worded items.

The finalised electronic survey was distributed along with an invitation to all members of the APCP. Respondents were also sought using social media (Twitter and Facebook) and via on-line professional discussion forums (i-CSP) in order to reach other neonatal physiotherapists who were not members of the APCP. Data collection was conducted using the UCL Opinio web-based survey tool, and all responses were anonymised at source. Data were transferred from the Opinio software into an Excel spreadsheet for storage, which allowed data transfer into Statistical Package for the Social Sciences (SPSS) software for statistical analysis. Descriptive statistics were used to report the findings. Furthermore, responses from the group of therapists who reported using outcome measures, were compared with those who reported not using them, using Mann-Whitney U tests for non-parametric data and presented as U and p-values. This was important, since one aim of the survey was to compare differences between users and non-users in their attitudes and approaches towards outcome measures. Where questions were binary (requiring a ‘yes’ or ‘no’ response), responses were compared using Fisher’s exact test after dividing the respondents into those who reported using outcome measures and those who reported not using them.

Results

A total of 91 responses were received, from which 43 were included in this study. Respondents of the 47 remaining questionnaires did not press the final ‘submit’ button, and their surveys were often substantially incomplete, so these were excluded. One other completed survey was excluded as the participant was not working in the UK. Each response came from a different hospital, so no hospital-specific focus of practice was over-represented. Of the included surveys, 79.1% (n=34) of respondents reported using developmental outcome measures on their units (Table 1).

Respondents were predominantly Band 7 physiotherapists (86%, n=37), working in units which had multiple bands of physiotherapy staff. All the units (100%, n=6) that included a Band 8 physiotherapist in their skill mix, used outcomes measures. This compares with 78% (n=29) of units where the highest level of experience was Band 7. Most of the respondents (44%) worked in units providing level 3 neonatal care, although 18 physiotherapists

worked across more than one level of care. 90% of physiotherapists who worked on a unit that included level 3 care, used outcome measures. This compares with 67% of those whose highest level of care was level 2. Neither band of physiotherapists present on units nor level of care significantly influenced the use of outcome measures on the unit (U=132.5, p=0.513 and U=110.5, p=0.177 respectively).

Of the 43 units included in the study, 58.1% (n=25) had input from a neonatal-specialist physiotherapist. ‘Neonatal-specialist’ was a self-reported term to differentiate from physiotherapists who would normally work with either paediatrics or adult patients. Where a neonatal-specialist physiotherapist was involved on the neonatal unit, 92% (n=23/25) of the respondents reported using outcome measures (Table 1). The presence of a neonatal-specialist on the unit was significantly related to the use of outcome measures (Fisher’s exact test: p=0.023).

Nearly all the respondents (n=40/43) participated in continuing professional development related to neonatal care. This included self-directed learning (90.7%) and peer learning and support within each NHS trust (81.4%) as part of their specialist training. 53% of physiotherapists also took part in postgraduate education with a neonatal component. The number of continuing professional development opportunities, training and support sources accessed by physiotherapists was significantly higher in the group who reported using outcome measures (U=71.5, p=0.011).

Current use of outcome measures

The most frequently utilised outcome measures were the Hammersmith Neonatal/Infant Neurological Examination (HNNE/HINE), the Lacey Assessment of Preterm Infants (LAPI) and the Prechtl’s Method of Qualitative Assessment of General Movements (GMs) (Table 2). Physiotherapists with greater years of experience in the use of developmental outcome measures, or who received specific training in their use, were significantly more likely to utilise a measure on their unit (Table 1).

Facilitators to using developmental outcome measures

The majority of respondents (n=39/43, 91%) had a positive attitude towards the use of outcome measures in clinical practice (item 8, ‘I feel that I have a positive attitude towards the use of developmental outcome measures’). The same number of respondents recognised the utility of outcome measures in providing insight for parents into their child’s physical functioning (item 21) (Table 3). Indeed, four physiotherapists specified in the ‘comments’ section that the use of outcome measures gave them the opportunity to have a discussion with the parents about different aspects of infant development,


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developmental support and the function of follow up clinics.

Barriers to using developmental outcome measures

Finding the opportune moment for completing a developmental assessment (item 18) was challenging for most of the physiotherapists (81.4%). Funding issues were reported by six respondents and were confirmed by 86.1% (n=37/43) of physiotherapists who considered the costs related to outcome measure acquisition (such as courses and materials) (item 16) an obstacle to using them, indicating the main barrier to their implementation (Table 3).

Differences in perceived barriers and facilitators between respondents using, and not using, developmental outcome measures on units

There were some significant differences between those respondents who used outcome measures and those who did not. There were significantly different responses to the statements ‘the use of a developmental outcome measure fits into my way of working at my unit’ (item 9) (U=18.5, p<0.001), and ‘I regard the use of a developmental outcome measure to be a good starting point for my physiotherapy interventions and for further referrals’ (item 10) (U=46, p<0.001). Those who used outcome measures tended to agree with these statements (with the exception of one respondent), whereas those who didn’t tended to disagree or be neutral (Table 3).

Regarding the outcome measures’ adaptability to the individual needs of infants (item 20), there was a significant difference between the two groups (U=79.5, p=0.016). Although 73.5% of the physiotherapists who utilised outcome measures agreed with the statement (n=25/34), one reported the difficulty of using non-completed data when transferring patients between units and for longitudinal data collection.

With regards to the environment, physiotherapists who had support from fellow physiotherapists (item 22) and other professionals (item 23) were significantly more likely to utilise outcome measures than those who did not (with U=34, p<0.001 and U=70, p=0.005 respectively) (Table 3).

Discussion

Existing guidelines recognise the need for neonatal physiotherapists to provide surveillance assessments and developmental support to neonates, from birth to the age of two years (NICE, 2017; CSP, 2017). From the results gained, it is encouraging that developmental outcome measures were utilised by 79% of the 43 respondents to the survey, facilitating the majority of the surveyed physiotherapists to provide a good-quality physiotherapy service and appropriate follow-up (Grol and Grimshaw, 2003; King et al.,

2011). The most frequently used outcome measures were HNNE/HINE, GMs and LAPI. This study found that the presence of a band 8 clinical specialist physiotherapist and/or input from a neonatal specialist physiotherapist on the unit, were significant indicators for the use of developmental outcome measures. Those physiotherapists who engaged with a variety of continuing professional developmental opportunities from a number of different sources were also more likely to use outcome measures in clinical practice.

The most representative neonatal service care was level 3 (44%). Although there was no significant relationship between the level of care and the use of outcome measures, most physiotherapists working on a unit that included level 3 care used outcome measures (89.5%). This suggests that outcome measures hold particular clinical importance for infants with more complex conditions, who require closer monitoring and tend to have a longer length of stay; thus, greater priority may be given to using outcome measures to monitor these infants.

The band of physiotherapy staff most present on units was Band 7 (86%), which is aligned with the previous findings (72.8% n=43/59, without missing data), (Ronan and Barron, 2012). Again, despite no significant relationship being found between the band of physiotherapists present on the unit and outcome measures utilisation, all units that included a Band 8 physiotherapist used outcome measures. This, combined with the findings regarding the use of outcome measures and involvement of a neonatal-specialist physiotherapist on the neonatal unit (Fisher exact test: p=0.023), suggests that the presence of a clinical specialist results in close monitoring of neonates.

Level and amount of training and support sources had a significant influence in outcome measure utilisation (higher levels of training and multiple training sources being related to outcome measure usage). These findings are in keeping with previous studies (Jette et al., 2009; Wedge et al., 2012). However, previous studies also argued that physiotherapists with greater clinical expertise tended to use informal outcome measures, relying on their own judgement and experience, whilst newly graduated physiotherapists were more familiar with integrating standardised outcome measures into their practice (King et al., 2011; Wedge et al., 2012). In contrast to these findings, in the current study, the neonatal physiotherapy role is an advanced clinical sub-speciality that requires relevant postgraduate education and appropriate professional experience (Brady and Smith, 2015). Physiotherapists with greater years of experience of using the tools, were more likely to implement standardised outcome measures in neonatal care. In addition, there is a difference between using a developmental outcome


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measure to track or anticipate changes over time, and using a short-term outcome measure to evaluate the effectiveness of a single treatment or course of treatments. As a result, the use of ‘informal outcomes’ by expert practitioners in other non-neonatal specialties may reflect the use of the outcome tools for a different purpose. In neonatal care, the tracking of development over a longer period may demand a standardised approach.

The most frequently used outcome measures were HNNE/HINE, GMs and LAPI. These tests, which have a discriminative and predictive function, evaluate posture, tone and the quality of movements in premature infants. The main differences are related to the administration time, infant handling involvement, equipment and training requirements (Kant, 2013). The tendency of utilising more than two outcome measures (67.7%, n=23/34), confirms the need for further studies to evaluate whether the concurrent use of those tools would increase the ability to detect or discriminate typically developing preterm infants to ensure the resources are targeted. In addition, size of caseload or local trust policies could be influencing factors in outcome measure preference and frequency of use, since Ronan and Barron (2012) identified that 39% of level 2 and 3 units (n=22/57, excluding 13 units that did not offer a neonatal physiotherapy service) had an ad hoc service, which was offered after a direct referral only.

The overall positive attitude towards the use of outcome measures (39/43), and the strong consensus regarding the utility of outcome measures to provide insight for parents into their child’s physical functioning (39/43), differed from findings by King et al. (2011). Those authors suggested that paediatric physiotherapists perceived the utilisation of outcome measures to be less important to parents. King et al. (2011) undertook a thorough literature review regarding barriers and facilitators to outcome measure use in paediatric physiotherapy, summarising that the unwillingness of physiotherapists to utilise standardised measurements may be related to the incapacity of those tools to detect small changes in children with physical and developmental disabilities. Thus, they preferred an individualised approach that celebrated small achievements in order to maintain the childrens’ motivation and parents’ appreciation for long-term therapies. These differences between paediatric and neonatal fields might be related to the type of condition and care. Although both fields adopt a family-centred approach, particular attention has been directed to parent-child interactions, parent education and early parent-administered physiotherapy on neonatal units in recent years (Ustad et al., 2016; Sweeney et al., 2009; Sweeney et al., 2010). In addition, the predictive nature of developmental tools might assume particular importance to parents

at this early stage.

High costs were described as significant limitations to the use of outcome measures on neonatal units, which reflected former findings from other physiotherapy specialist areas including musculoskeletal, neurological and paediatric fields (Jette et al., 2009; Wedge et al., 2012; Van Peppen et al., 2008; King et al., 2011). Moreover, finding the opportune moment for outcome measure use was perceived as a barrier by the majority of respondents (81.4%). From the literature, it seems that this limitation is specific to the neonatal setting and might partially explain the physiotherapists’ tendency to integrate different tools, some of which involve purely observational assessment, into the neonatal assessment.

Significant differences were found between those physiotherapists that used outcome measures and those that did not. In particular, there was strong consensus among respondents who used outcome measures that they ‘fit into the way of working on units’ (94.1%) and are a ‘starting point for physiotherapy intervention and further referrals’ (94.1%).

There were also significant differences between respondents using outcome measures and those not, in terms of the level of support offered by fellow physiotherapists and other professionals. Those respondents using outcome measures felt significantly more supported by their colleagues and peers, (p<0.001 and p=0.005 for therapists and other professionals respectively). As opposed to former studies, which identified culture and organisation as determining factors in outcome measure implementation (Jette et al., 2009; King et al., 2001; Wedge et al., 2012), interdisciplinary support assumed significant importance in the neonatal physiotherapy field. This suggests that where a supportive environment exists, physiotherapists feel comfortable utilising outcome measures.

It is acknowledged that this survey has several limitations. The non-standardised term, ‘developmental outcome measure’, was utilised to group different neurological, behavioural or motor performance tests, since they assess various aspects of neonatal development and they were designed for serial/longitudinal use to track the developmental evolution of infants (Brazelton and Nugent, 2011; Dubowitz et al., 1999; Einspieler, 2004, Lacey, 2004). The lack of consensus regarding this terminology may have impacted on the interpretation of questions and statements, leading to ambiguity of findings. There were a significant number of non-responses to certain individual items, which could have impacted on the validity of findings. These may have been reduced with a longer piloting process. However, the study was undertaken within the context of a fairly limited


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time-frame and the findings remain a useful addition to the literature. The use of an electronic survey may explain some of the problems in completeness, consistency and a lower response rate compared to the epistolary method (Bethlehem, 2009; Ritter et al., 2004; Sivo et al., 2006). However, given that neonatal physiotherapy is a fairly small subspecialty, the response rate was deemed to be sufficient and representative of this group.

Conclusion

The results of this study suggest that outcome measures are commonly used by neonatal physiotherapists in the UK. The most frequently used outcome measures were HNNE/HINE, GMs and LAPI. There was generally a positive attitude towards outcome measure usage, but with some barriers to their implementation including the high cost. The non-use of outcome measures could be attributed to the level of expertise, type of training and a lack of interdisciplinary support. This provides a useful discussion point for experts in the field, when considering the impact that staff development and support could have on outcome measure utilisation in the future.

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Ritter, P., Lorig, K., Laurent, D. & Matthews, K. (2004). Internet versus mailed questionnaires: a randomized comparison. Journal of Medical Internet Research, 6.

Ronan, C. & Barron, J. (2012). National benchmarking of neonatal physiotherapy services. APCP Journal, 3, 19-26.

Santhakumaran S., Statnikov, Y., Gray, D., Battersby, C., Ashby, D. & Modi, N., (2017). Survival of very preterm infants admitted to neonatal care in England 2008–2014: time trends and regional variation. Archives of Disease in Childhood - Fetal and Neonatal Edition, F1 – F8.

Sivo, S. A., Saunders, C., Chang, Q. & Jiang, J. J. (2006). How low should you go? Low response rates and the validity of inference in IS questionnaire research. Journal of the Association for Information Systems, 7, 17.

van Sonderen, E., Sanderman, R. & Coyne, J. (2013). Ineffectiveness of Reverse Wording of Questionnaire Items: Let’s Learn from Cows in the Rain. PloS One 8(7): e68967.

Swinkels, R., van Peppen R., Wittink, H., Custers, J. & Beurskens, A. (2016). Current use and barriers and facilitators for implementation of standardised measures in physical therapy in the Netherlands. BMC Musculoskeletal Disorders 2011; 12: 106.

Sweeney, J. K., Heriza, C. B. & Blanchard, Y. (2009). Neonatal physical therapy. Part I: clinical competencies and neonatal intensive care unit clinical training models. Pediatric Physical Therapy, 21, 296-307.

Sweeney, J. K., Heriza, C. B., Blanchard, Y. & Dusing, S. C. (2010). Neonatal physical therapy. Part II: Practice frameworks and evidence-based practice guidelines. Pediatric Physical Therapy, 22, 2-16.

Ustad, T., Evensen, K. A. I., Campbell, S. K., Girolami, G. L., Helbostad, J., Jorgensen, L., Kaaresen, P. I. & Øberg, G. K. (2016(. Early parent-administered physical therapy for preterm infants: a randomized controlled trial. Pediatrics, e20160271.

Van der Wees, P. J., Zagers, C. A., De Die, S. E., Hendriks, E. J., Nijhuis-Van Der Sanden, M. W. & De Bie, R. A. (2013). Developing a questionnaire to identify perceived barriers for implementing the Dutch physical therapy COPD clinical practice guideline. BMC health services research, 13, 159.

Van Peppen, R. P., Maissan, F. J., Van Genderen, F. R., Van Dolder, R. & Van Meeteren, N. L. (2008). Outcome measures in physiotherapy management of patients with stroke: a survey into self-reported use,

and barriers to and facilitators for use. Physiotherapy Research International, 13, 255-270.

Wedge, F. M., Braswell-Christy, J., Brown, C. J., Foley, K. T., Graham, C. & Shaw, S. (2012). Factors influencing the use of outcome measures in physical therapy practice. Physiotherapy theory and practice, 28, 119-133.


20


Tables

Table 1: Summary of participants

Use of OMs (YES) n=34 (79%)

Non-use of OMs (NO) n=9 (21%)

Total n=43 (100%) Significance

Highest level of neonatal care provided

123OtherNot reported

4 (12%)8 (24%)17 (50%)1 (3%)4 (12%)

04 (44%)2 (22%)1 (11%)2 (22%)

4 (9%)12 (28%)19 (44%)2 (5%)6 (14%)

U = 110.5p = 0.177

Physiotherapy band levels present on the neonatal unit

Band 6Band 7Band 8Bands 6 & 7Bands 6 & 8Bands 7 & 8Bands 5, 6 & 7Bands 4, 6, 7 & 8

1 (3%)15 (44%)3 (9%)12 (35%)1 (3%)1 (3%)01 (3%)

1 (11%)2 (22%)05 (56%)001 (11%)0

2 (5%)17 (40%)3 (7%)17 (40%)1 (2%)1 (2%)1 (2%)1 (2%)

U = 132.5p = 0.513

Number of years using developmental outcome measures

no years< 2 years2-5 years> 5 years

06 (18%)9 (26%)19 (56%)

3 (33%)2 (22%)1 (11%)3 (33%)

3 (7%)8 (19%)10 (23%)22 (51%)

U = 90p = 0.041

Training received in the use of developmental outcome measures

YesNo

30 (88%)4 (12%)

3 (33%)6 (67%)

33 (77%)10 (23%) p = 0.002

Whether therapists have read the APCP competence framework for working in neonatal care

YesNo

31 (91%)3 (9%)

8 (89%)1 (11%)

39 (91%)4 (9%)

p = 1.00

OMs: developmental outcome measures; APCP: Association for Paediatric Chartered Physiotherapists; Statistical Significance p<0.05; ‘Other’ refers to: n=1 ’unsure’; n=1 consultant level therapist

21

Table 2: Frequency of the use of developmental outcome measures

OMs Nevern (%)

<5 timesn (%)

5-10 timesn (%)

11-25 timesn (%)

>25 timesn (%)

Response ratea

n (%)

HNNE/HINEGMs LAPINBASNIDCAPTIMPNAPIOthers: AIMSOthers: Bayley

4 (11.8%)5 (14.7%)8 (23.5%)11 (32.4%)17 (50%)17 (50%)19 (55.9%)00

4 (11.8%)3 (8.8%)2 (5.9%)7 (20.6%)01 (3%)00 2 (5.9%)

4 (11.8%)1 (3%)1 (3%)1 (3%)00000

4 (11.8%)6 (17.6%)1 (3%)01 (3%)0000

12 (35.3%)12 (35.3%)14 (41.2%)3 (8.8%)2 (5.9%)005 (14.7%)1 (3%)

28 (82.4%)27 (79.4%)26 (76.5%) 22 (64.7%) 20 (58.8%)18 (53%) 19 (55.9%) 5 (14.7%) 3 (8.8%)

OMs: developmental outcome measures; HNNE/HINE: Hammersmith Neonatal Neurological Examination/Hammersmith Infant Neurological Examination; GMs: Prechtl’s Method of Qualitative Assessment of General Movements; LAPI: Lacey Assessment of Preterm Infants; NBAS: Brazelton Neonatal Behavioural Assessment Scale; NIDCAP: Newborn Individualised Care and Assessment Program; TIMP: Test of Infant Motor Performance; NAPI: Neurobehavioural Assessment of the Preterm Infant; AIMS: Alberta Infant Motor Scale; Bayley: Bayley Scales of Infant and Toddler Developmenta [Total number of responses: n=34]

Table 3: Summary of barriers and facilitators questionnaire section

Items Use of OMs

Strongly disagreen (%)

Disagree

n (%)

Do not agree nor disagreen (%)

Agree

n (%)

Strong-ly agreen (%)

Total

n (%)

Mann-Whitney U testp-value

8. I feel that I have a positive attitude towards the use of developmental outcome measures[Barrier (1) → Facilitator (5)]

Yes

No

2 (5.9%)

0

0

0

02 (22.2%)

14 (41.2%)

4 (44.5%)

18 (53%)

3 (33.3%)

34 (100%)

9 (100%)

U = 115p = 0.208

9. The use of a developmental outcome measure fits into my way of working at my unit[Barrier (1) → Facilitator (5)]

Yes

No

1 (3%)

0

0

4 (44.5%)

1 (3%)

4 (44.5%)

13 (38.2%)

1 (11.1%)

19 (55.9%)

0

34 (100%)

9 (100%)

U = 18.5p < 0.001

10. I regard the use of a developmental outcome measure to be a good starting point for my physiotherapy interventions and for further referrals[Barrier (1) → Facilitator (5)]

Yes

No

1 (3%)

0

0

0

1 (3%)

6 (66.7%)

10 (29.4%)

2 (22.2%)

22 (64.7%)

1 (11.1%)

34 (100%)

9 (100%)

U = 46p < 0.001


22

11. It is important that developmental outcome measures can be used before 38-40 weeks postmenstrual age[Barrier (1) → Facilitator (5)]

Yes

No

1 (3%)

0

3 (8.8%)

1 (11.1%)

3 (8.8%)

2 (22.2%)

16 (47.1%)

5 (55.6%)

11 (32.4%)

1 (11.1%)

34 (100%)

9 (100%)

U = 117p = 0.246

12. I consider that a complex layout of a developmental outcome measure can be an obstacle to using it[Facilitator (5) ← Barrier (1)]

Yes

No

0

0

4 (11.8%)

0

4 (11.8%)

2 (22.2%)

19 (55.9%)

5 (55.6%)

7 (20.6%)

2 (22.2%)

34 (100%)

9 (100%)

U = 145.5p = 0.804

13. The need for additional equipment (such as a video camera) can be an obstacle to using the developmental outcome measure in my unit[Facilitator (5) ← Barrier (1)]

Yes

No

1 (3%)

0

6 (17.6%)

1 (11.1%)

4 (11.8%)0

14 (41.2%)

5 (55.6%)

9 (26.5%)

3 (33.3%)

34 (100%)

9 (100%)

U = 123.5p = 0.35

14. The number of items that require infant handling can be an obstacle to using a developmental outcome measure[Facilitator (5) ← Barrier (1)]

Yes

No

0

0

6 (17.6%)

3 (33.3%)

2 (5.9%)1 (11.1%)

16 (47.1%)

5 (55.6%)

10 (29.4%)

0

34 (100%)

9 (100%)

U = 96p = 0.067

15. I feel that the use of developmental outcome measures can be too time-consuming[Facilitator (5) ← Barrier (1)]

Yes

No

6 (17.6%)0

17 (50%)4 (44.5%)

3 (8.8%)

2 (22.2%)

6 (17.6%)2 (22.2%)

2 (5.9%)1 (11.1%)

34 (100%)9 (100%)

U = 108p = 0.15

16. The cost of acquiring certain developmental outcome measures (such as courses and materials) can be an obstacle to using them[Facilitator (5) ← Barrier (1)]

Yes

No

1 (3%)

0

1 (3%)

0

3 (8.8%)

1 (11.1%)

20 (58.8%)

4 (44.5%)

9 (26.5%)

4 (44.5%)

34 (100%)

9 (100%)

U = 124.5p = 0.341

17. It is difficult to understand the use of developmental outcome measures in patients who seem healthy[Facilitator (5) ← Barrier (1)]

Yes

No

12 (35.3%)

1 (11.1%)

16 (47.1%)

6 (66.7%)

4 (11.8%)

2 (22.2%)

1 (3%)

0

1 (3%)

0

34 (100%)

9 (100%)

U = 120p = 0.281

18. Finding the right moment (such as infant awake) for the developmental assessment is often challenging[Facilitator (5) ← Barrier (1)]

Yes

No

0

0

5 (14.7%)

0

1 (3%)

2 (22.2%)

22 (64.7%)

5 (55.6%)

6 (17.7%)

2 (22.2%)

34 (100%)

9 (100%)

U = 148p = 0.863


23

19. The use of developmental outcome measures in neonatal units is too stressful for the infant[Facilitator (5) ← Barrier (1)]

Yes

No

5 (14.7%)

0

19 (55.9%)

5 (55.6%)

5 (14.7%)

4 (44.5%)

3 (8.8%)

0

2 (5.9%)

0

34 (100%)

9 (100%)

U = 127.5p = 0.399

20. Developmental outcome measures can be adapted to the individual needs of the infants[Barrier (1) → Facilitator (5)]

Yes

No

0

0

3 (8.8%)

3 (33.3%)

6 (17.7%)

3 (33.3%)

20 (58.8%)3 (33.3%)

5 (14.7%)0

34 (100%)9 (100%)

U = 79.5p = 0.016

21. Developmental outcome measures give parents an insight into their child’s physical functioning[Barrier (1) → Facilitator (5)]

Yes

No

0

0

1 (3%)

0

1 (3%)

2 (22.2%)

18 (53%)

6 (66.7%)

14 (41.2%)

1 (11.1%)

34 (100%)

9 (100%)

U = 96p = 0.055

22. Fellow neonatal physiotherapists cooperate in applying the developmental outcome measures[Barrier (1) → Facilitator (5)]

Yes

No

0

1 (11.1%)

0

1 (11.1%)

6 (17.7%)

6 (66.7%)

20 (58.8%)

1 (11.1%)

8 (23.5%)

0

34 (100%)

9 (100%)

U = 34p < 0.001

23. Other professionals support the use of developmental outcome measures[Barrier (1) → Facilitator (5)]

Yes

No

0

2 (22.2%)

0

1 (11.1%)

2 (5.9%)

2 (22.2%)

22 (64.7%)3 (33.3%)

10 (29.4%)1 (11.1%)

34 (100%)9 (100%)

U = 70p = 0.005

24. Managers/directors support the use of developmental outcome measures[Barrier (1) → Facilitator (5)]

Yes

No

0

2 (22.2%)

1 (3%)

0

7 (20.6%)

3 (33.3%)

17 (50%)3 (33.3%)

9 (26.5%)1 (11.1%)

34 (100%)9 (100%)

U = 92.5p = 0.053

OMs: developmental outcome measuresStatistical significance (p<0.05)


24


1. Do you currently use a developmental outcome measure on your unit?

□ Yes□ No: Please go to question number 4

2. How often have you used the following developmental outcome measures?If you use other developmental outcome measures, please specify in the text boxes

Never <5 times 5-10 times

11-25 times

>25 times

Hammersmith Neonatal/Infant Neurological Examination (Dubowitz or NANI)

□ □ □ □ □

Lacey Assessment of Preterm Infants (LAPI) □ □ □ □ □

Brazelton Neonatal Behavioural Assessment Scale (NBAS) □ □ □ □ □

Neurobehavioural Assessment of the preterm Infant (NAPI) □ □ □ □ □

Newborn Individualised Care and Assessment Program (NIDCAP) □ □ □ □ □

Prechtl’s Method of Qualitative Assessment of General Movements (GMs)

□ □ □ □ □

Test of Infant Motor Performance (TIMP) □ □ □ □ □

Other/further comments

3. What is the youngest postmenstrual age range (weeks) that you would assess a baby using a developmental outcome measure?

(please add additional comments in the box provided)

Please go to question number 5

□ 24-27 □ 28-29□ 30-31□ 32-34□ 35-37□ 38-40□ >40: Please Specify ………

Other/further comments:

4. If it were possible, which outcome measure/s would you like to introduce

(tick all that apply on the list)

□ Hammersmith Infant Neurological Examination (Dubowitz or NANI) □ Lacey Assessment of Preterm Infants (LAPI)□ Brazelton Neonatal Behavioural Assessment Scale (NBAS)□ Neurobehavioural Assessment of the preterm Infant (NAPI)□ Newborn Individualised Care and Assessment Program (NIDCAP)□ Prechtl’s Method of Qualitative Assessment of General Movements (GMs)□ Test of Infant Motor Performance (TIMP)□ I don’t know□ None□ Other: Please Specify ………


Appendix 1: Questionnaire

Developmental outcome measures

25

5. What is your level of experience with the use of developmental outcome measures

□ I have no experience□ <2 years of experience□ 2-5 years of experience□ >5 years of experience


6. Have you received training in the use of developmental outcome measures?

□ Yes□ No

Please state the outcome measure/s that you have been trained to use, and the type of training that you undertook (e.g. inservice training, APCP course, etc):


Barriers and facilitators to the implementation of tests of infant development

The Association of Paediatric Chartered Physiotherapists (APCP) published on 2011 and updated on 2015 “A Competence Framework and Evidenced-Based Practice Guidance for the Physiotherapist working in the Neonatal Intensive Care and Special Care Unit in the United Kingdom”, in which is recommended the use of developmental outcome measures.

Following are a couple of statements about the use of developmental outcome measures. We would like to know whether you agree with the statement or not and in what degree. If you do not have a strong opinion, please try to find out if it is more like ‘agree’ or more like ‘disagree’. If you really do not know, you can select the option ‘do not agree nor disagree’.

7. Which of the following statement is true? □ I read the APCP guidance□ I am aware of the APCP guidance, but I have not read them yet□ I am not aware of the APCP guidance

Strongly disagree

Disagree Do not agree nor disagree

Agree Strongly agree

8. I feel that I have a positive attitude towards the use of developmental outcome measures □ □ □ □ □

9. The use of developmental outcome measures fits into my way of working at my unit □ □ □ □ □

10. I regard the use of a developmental outcome measure to be a good starting point for my physiotherapy interventions and for further referrals


□ □ □ □ □

26


11. It is important that developmental outcome measures can be used before 38-40 weeks postmenstrual age


□ □ □ □ □

12. I consider that a complex lay-out of a developmental outcome measure can be an obstacle to using it


□ □ □ □ □

13. The need for additional equipment (such as a video camera) can be an obstacle to using the developmental outcome measure in my neonatal unit


□ □ □ □ □

14. The number of items that require infant handling can be an obstacle to using a developmental outcome measure


□ □ □ □ □

15. I feel that the use of developmental outcome measures can be too time-con-suming


□ □ □ □ □

16. The cost of acquiring certain devel-opmental outcome measures (such as courses and materials) can be an obsta-cle to using them


□ □ □ □ □

17. It is difficult to understand the use of developmental outcome measures in patients who seem healthy


□ □ □ □ □

18. Finding the right moment (such as infant awake) for the developmental assessment is often challenging


□ □ □ □ □

19. The use of developmental outcome measures in neonatal units are too stressful for the infant


□ □ □ □ □

20. Developmental outcome measures can be adapted to the individual needs of the infant


□ □ □ □ □

27

21. Developmental outcome measures give parents an insight into their child’s physical functioning


□ □ □ □ □

22. Fellow neonatal physiotherapists coop-erate in applying the developmental outcome measures


□ □ □ □ □

23. Other professionals support the use of developmental outcome measures


□ □ □ □ □

24. Managers/directors support the use of developmental outcome measures


□ □ □ □ □

25. What is the name of the hospital in which you work?

This question is to account for multiple responses from the same unit. The name of the unit will NOT be revealed in the analysis or dissemination of findings

How many beds does your neonatal unit have, and at what level are these beds?

26. Level 1:27. Level 2:28. Level 3:


29. What bands of physiotherapy staff work on the neonatal unit?

(tick all that apply)

□ Band 3 assistant□ Band 4 assistant / T.I□ Band 5□ Band 6□ Band 7□ Band 8

Please specify approximately how many hours per week your physiotherapy team spends on the neonatal unit:

30. Where does your physiotherapy input come from?


□ Neonatal-specialist physiotherapist□ Predominantly paediatric physiotherapists from

acute trust□ Predominantly adult or general physiotherapists

from acute trust□ Paediatric physiotherapists from community trust


Neonatal physiotherapy service provision and training Following are a couple of questions about your neonatal physiotherapy service and training.


28

31. What types of physiotherapy interven-tions are your neonatal physiothera-pists involved with?


□ Neuro-developmental evaluation □ Neuro-developmental interventions□ Respiratory / chest clearance□ Orthopaedic – e.g. OBPP, Talipes(Obstetric Brachial Plexus Palsy)□ Psychosocial meetings□ Neonatal follow-up clinics□ Parent’s education/collaboration


32. As a neonatal physiotherapist, what specialist training and support have you undertaken?


□ I am a member of APCP (Association of Paediatric Chartered Physiotherapists) neonatal group

□ I have attended courses related to neonatal care□ I have undertaken postgraduate education, which

included neonatal care□ I have been involved in peer learning and support

within the trust□ I have received teaching/training from senior col-

leagues□ I have undertaken my own self-directed learning□ Other (please state)


29


C. Ronan*, D. Coggings, D. Carr*Corresponding author: [email protected]

ABSTRACT

The paediatric Physiotherapy service at the Royal London Hospital, Bart’s Health NHS Trust, was reviewed in 2013 in order to provide the most effective service for its patients. Following this review, a new pattern of extended hours working was implemented and reviewed at regular intervals for three years.

This paper details how the need for service re-design was established, and the change management process that ensued.

Following implementation, an increase in productivity was demonstrated by 95 (55-125) patients per month being seen on weekday evenings, and 134 orthopaedic day case patients being seen and discharged out of hours per annum. The overnight call out rate dropped by 38%, with less calls pre 03:30am. Staff sickness was lower, and compensatory rest was assured for staff.

Initial expenditure on a band 6 physiotherapist to enable this pattern of working was offset by reduced on on-call and weekend pay, and bed day savings from day-case patients being discharged more effectively. At the three year review, the new pattern was shown to have a cost saving of over £15,000 despite increasing the establishment.

The extended working pattern has been shown to be a cost effective way of working within an acute Tertiary Paediatric Physiotherapy service.

Introduction

The paediatric physiotherapy services at The Royal London Hospital (RLH), Bart’s Health NHS Trust, provide tertiary level care for paediatric orthopaedic, neurology, medical and surgical specialties, including intensive care, cystic fibrosis, neonatology, gastroenterology and elective orthopaedics. Additionally, the Trust incorporates a busy A&E with a helicopter emergency service. This provides regular paediatric trauma admissions who journey through the Trust from A&E to intensive care, rehabilitation and home. In 2011, many of the adult physiotherapy services moved to a seven day working model in response to a Trust incentive to provide higher quality care at a lower cost. In 2013, the Paediatric Physiotherapy Service at RLH was reviewed to establish the most appropriate model of extended hours care.

The following paper summarises the PEST model of analysis of the previous 08:30 – 16:45 working day and on call service, followed by the options considered and implementation of the new system. This was evaluated at regular intervals for three years post implementation with consideration of service delivery and financial implication.

The Need for Change

A ‘PEST analysis’, or consideration of the Political, Economic, Social and Economic factors impacting

on a service, was chosen as the model for service evaluation due to its consideration of many different aspects of the macro environment (Businessballs, 2010). By understanding the many different angles which impact on a need for change, a thorough review can be undertaken, however this only provides an accurate analysis at the time it was undertaken, which in this review was summer 2013.

Political

Prior to this review being undertaken, there had been strong interest in improving access to services across seven days, initially raised onto the political agenda by Everyone Counts: planning for patients 2013/14 (NHS Commissioning Board, 2013).

The focus on seven day services was health outcome driven, as highlighted by Equity and Excellence: Liberating the NHS (Department of Health, 2010). Improvements in paediatric physiotherapy service provision were strongly linked with the health outcome domains of ‘helping people to recover from episodes of ill health or following injury’ (domain 3) and ‘improving children and young people’s experience of healthcare’ (domain 4) (NHS outcome frameworks for 2013/14, DOH, 2012).

Large scale pilots of extended hours of working had also been carried out prior to this service review with demonstrated improvement including; reduced time to initial assessment (Hunt, 2009), improvements in

C. Ronan / APCP Journal Volume 9 Number 1 (2018)

30


adherence to national standards and reduction in number of ventilator days (NHS Improvement, 2011). Additionally, patient satisfaction was reported as very high in Trusts which had implemented seven-day working (NHS improvements, 2011).

Inequalities of care were also evident in current healthcare provision. While Securing Excellence in Commissioning Primary Care (NHS commissioning Board, 2012a) and Equity and Excellence: Liberating the NHS (Department of Health, 2010) both highlighted an ambition to reduce inequality, the Equality Act (UK Parliament, 2010) gave the NHS a clear opportunity to eliminate discrimination and work towards standardised services. At the time of this service review, adult services within this Trust had implemented a level 3 seven-day service (NHS improvement, 2011), while paediatric services remained at a level 1 (Figure 1). Age discrimination is one of the nine ‘protected characteristics’ for which discrimination is stipulated against by this act, with the Equality Delivery System (EDS) (NHS, 2012) giving the NHS a tool to identify specific objectives in order to comply with the Equality Act (UK Parliament, 2010). Objectives which address these inequalities should then be incorporated into mainstream business planning, as was the case for service reform following this PEST analysis.

Economic

In 2013, the NHS was facing its tightest budget for the last 50 years (Nuffield Trust, 2012). Service development considerations which improved productivity at a cost-neutral or cost-saving level therefore became priorities at this time to address the projected funding gap of £44 billion by 2021/22 (Nuffield Trust, 2012).

To prevent dilution of the current service, it was

acknowledged that there would be initial financial outlay in terms of staffing in order to introduce an extended pattern of working (Chartered Society of Physiotherapy, 2008; Hunt, 2009; NHS Improvement, 2011). It was anticipated that cost avoidances, in terms of reduction in bed days and on call costs would offset these costs, improving the economic attractiveness of this option.

While pilot data from a paediatric seven-day service was not available, bed day savings were identified from adult pilots, particularly in terms of reductions in bed days. Such cost avoidances are not specific to adult patients, and it was anticipated that extended evening and weekend hours would facilitate paediatric orthopedic discharges within this Trust. An internal review of orthopedic delayed discharges in 2013 demonstrated that two patients a week were delayed discharges resulting from lack of physiotherapy access on an evening or weekend, especially following day case surgery. Based on an average bed cost of £273 per day (NHS institute for Innovation and Improvement, 2013), these financial savings were projected to be around £23,400 per annum.

Cost benefits had not only been demonstrated in terms of reduced bed days. Following introduction of a seven-day working model to the cardio-respiratory service, Guy’s and St Thomas’ NHS Foundation Trust demonstrated a reduction in out of hour physiotherapy call outs as a result of enhanced care over longer hours (NHS Improvement, 2011). Incorporating extended hours within a current 37.5 hour week removes a proportion of on-call costs from the budget as services are re-structured.

Review of on-call data for Bart’s health for the year preceding the review, 363 on-call hours had taken place between the end of the working day and 21:30 at night. Paid at 1.5x time, at a mid-point band 6 this equated to £9,040 of on-call costs. Additionally, hours worked at weekends which were paid at overtime, equating to around £18,000 per annum. This total of over £27,000 could potentially be incorporated in standard working hours, offsetting the cost of initial staffing outlay.

Social

Social factors within the local catchment area for the Trust were considered to have a direct implication on the services the local population required. Levels of general child health in the boroughs surrounding the Trust were well below average (Tower Hamlets, 2012), with high levels of childhood disability (Department for Children, Schools and Families, 2008). While a reflection of local socioeconomic status and high levels of consanguinity within the local population (Department for Children, Schools and Families,

Level 1:Some extended hours, limited

weekend access

Level 2: Services 7 day per week, not

necessarily the same access each day

Level 3: Integrated 7 day service involving

several specialities

Level 4: Integrated 7 day service across a

whole system

The four levels of Seven Day working (NHS Improvement, 2011)

Escalatin

g 7-day service m

odels

Current Paediatric Physiotherapy Service

Current Adult Physiotherapy Service

Figure 1: The four levels of Seven Day working and Paediatric / Adult inequality within Bart’s Health NHS Trust in 2012

31

2008), many of these children have complex health needs and regularly present to Tertiary services.

Additionally, the local population was found to have a rising level of acute hospital admissions due to accidents and trauma while the national average was falling (Department for Children, Schools and Families, 2008). As the Trust has a Paediatric A&E department as well as a helicopter emergency service, admissions for Paediatric Trauma are common. Paediatric physiotherapists play an important role in discharging patients, freeing up beds for acute admissions and avoiding breaches in waiting times in A&E. Access to these services over extended hours, seven days a week would assist with bed flow for patients.

Technological

With a PEST analysis, some factors have more impact on service evaluation and change than others. Within this review, the political, economic and social factors were heavily weighted forces which drove this service change towards a seven-day model. Technology has progressed to allow patients unlimited and easy access to a wide range of information surrounding their care. This has created many opportunities for patient awareness and empowerment and expectations on services can be heightened. While meeting patient expectations is high on the NHS political agenda (Department of Health, 2010; Department of health 2012), Trusts are facing increasing difficulties with meeting patients expectations and achieving adequate levels of satisfaction. Improvements in patient satisfaction as a result of seven-day services have been demonstrated by national pilots (NHS Improvement, 2011). Responding to initiatives which improve patient experience in line with changing expectations (such as seven day working) will lead to success in terms of political drivers, satisfaction and service reputation.

Service Redesign

Following the PEST analysis, a clear requirement for service re-design was identified, which needed to enhance productivity on a cost neutral (or better) basis.

The existing service in 2013 provided full access to paediatric physiotherapy services Monday – Friday 08:30 – 16:45. Weekday evenings were covered on-call. At a weekend, there was a ‘first on’ and a ‘second on’ physiotherapist for each day of the weekend, with the overnight on call being covered by the ‘first on’ physiotherapist when the day work was completed. Only respiratory patients could access services out of standard working hours. An evaluation of hours worked during the weekend days demonstrated an average requirement of 7.5hrs of respiratory physiotherapy per weekend day, and 712 hours of overnight on-call work per annum.

Following this review, the case for service redesign was presented to the Trust. Three options based on ‘sample rotas’ were identified:

1) Continue with the same staffing, resulting in no ability to provide extended working hours and no ability to provide compensatory rest for staff

2) Increase staffing by 1 WTE to move to an extended working pattern but not full seven day working (projected to be cost neutral)

3) Increase staffing by 4 WTE to move to full shift seven day working

Based on the requirement for a projected cost neutral service, the Trust subsequently authorised option 2 - an increase in the establishment of 1WTE band 6 physiotherapist to enable extended working hours.

Change Management

In the public sector, pressures to change services are predominantly instigated by cost cutting and rationalisation of services (Shanley, 2007). As such, emphasis can be greatly shifted towards economic factors, with personal and socio-dynamic factors being neglected (Shanley, 2007; Suc, Prokosch, & Ganslandt, 2009). Staff fear of change is based on the implications of shift working on their personal lives and working conditions. Physiotherapy staff are a high impact stakeholder for seven-day working, with moderate to high power over the change especially when applying group pressure (NHS Institute for Innovation and Improvement, 2008). It is therefore understandable that change is postulated to fail if personnel factors, in this case physiotherapy staff fears, are not adequately addressed (Kuokkanen et al., 2007; Mustain et al., 2008; Suc et al., 2009). The aim of initial change management during implementation of this pattern of working was to ensure that the majority of staff were accepting of change. The Trust agreed to increase the establishment. Decision-making was delegated to the physiotherapy team, to ensure transparency and ownership. This model had previously been reported by Suc et al. (2009), where a nursing team underwent successful transformational change.

Following a meeting to clarify the need for change, all members of staff were asked to draw up any realistic options they felt would incorporate:

1) Cover until after 22:00 on weekdays (as peak on call time was 21:00-22:00)

2) Weekends and evening shifts to be incorporated in a 37.5 hour working week

3) Is feasible with only a 1 WTE increase in establishment

Three workable options were received, and the staff


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entered a consultation period on the proposed change, overseen by HR and CSP representation.

The requirement to incorporate weekend and evening hours into the standard 37.5 hour working week provided a unique logistical challenge when considering staff contracted by Barts Health NHS Trust to carry out on call but currently on rotation outside the main hospital base. Because of this, a small team of 4 therapists would provide cover which was still paid as overtime, and this was felt to be best allocated to covering ‘short shifts’ on weekend mornings. All ‘late shifts’ at weekends and evenings would be carried out on a rota by the remaining 12 members of staff and incorporated into the standard 37.5hour working week with a day off in lieu the following week after working a weekend (Figure 2)

For staff working ‘late shifts’ they elected to work five consecutive late shifts 13:45-22:00, Monday-Friday once every 12 weeks, and cover the linking on calls. These physiotherapists had the ability to come in late at 16:30 if they had had call outs overnight, to ensure adequate compensatory rest, i.e. on ‘busy’ weeks this would move towards working a night shift pattern. The additional member of staff funded to enable this pattern of working ‘backfilled’ the person on lates, covering their normal role. Weekend late shifts were worked on a rota as single shifts.

At six months post implementation, feedback was received that five days of consecutive lates was having an adverse effect on work life balance. Again, staff were given options and elected to move to three consecutive late shifts – Sunday to Tuesday;

and Wednesday to Friday, with a single shift on a Saturday. Although this increased the frequency of the duties, this improved satisfaction and this pattern was maintained for the remainder of the three years of this review. Over the period of implementation, one member of staff (who had not previously worked on calls) left the Trust citing this as a contributory factor. There were no other issues with recruitment and retention or ongoing issues with staff satisfaction.

The New Pattern of Working: Evaluation

The new pattern of working as described above was implemented in summer 2014. As the main aims of this service re design were improvements in service delivery in a cost neutral way, the structure was evaluated at regular intervals up to three years post implementation.

Service Delivery

Over the three-year review period, 95 patients per month were seen on average during a weekday late shift (range 55-125). This equates to an average of 5 treatments per weekday evening.

The breakdown of the location of patients treated was 42% Paediatric Critical Care Unit (PCCU); 30% Cystic Fibrosis (CF) wards; 15% general medicine; 8% orthopaedic discharges and 5% neonatal unit.

1) Impact on staff sickness

During the implementation of the new pattern of working, concerns were raised by staff regarding the potential impact on sickness that may occur if people


'Old' pattern of working 'New' pattern of working

On call cover 16:45 - 08:3022:00 - 08:30 Weekdays20:00 - 08:30 Weekends

Weekday EveningsNo cover except emergency respiratory on call cover

Cover provided for any appropriate patients (respiratory, orthopaedic discharges) until 10pm at night

Weekend coverCover provided for as long as it took to see priority respiratory patients

Full cover provided between 08:30 and 20:00 - any appropriate patients seen

Structure of weekend cover

Average of 7.5 hours a day worked by a 'first on' (and 'second on' if needed) physiotherapist who were paid full overtime for the hours worked

08:30 - 12:00: Worked by staff on rotation outside the main Hospital and paid full overtime 11:45 - 20:00: Worked by all staff in the main hospital as part of the 37.5 hour working week

Figure 2: Comparison of the ‘old’ and ‘new’ out of hours structure

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became overtired.

Sickness rates were analysed using data gained from an electronic absence monitoring system used within the Trust. Comparison of the sickness data for the year before and the year after implementation showed no increase in sickness rates, dropping from 5.78 days to 4.82 days per member of staff doing on-calls during a one year period.

2) Impact on overnight call-out rates and time of call outs

Prior to implementation of the new structure, it was anticipated that overnight call-out rates would reduce secondary to an extended evening service.

Data was retrieved from an electronic on-call record, in which accuracy had been ensured for many years by not authorising on-call payment to staff unless the record had been completed. The electronic data for the year after implementation was compared with the preceding three years, as demonstrated in Table 1 and Figure 3. This demonstrates the yearly increase in call outs as PCCU increased the acuity of its work, and the subsequent dramatic drop in on-call rates following implementation of the new structure, which has been maintained over time. Of importance to the financial implication of this structure, the number of hours claimed as on call overtime by the team reduced from 712 hours in 2013/14, to 224 hours in 2014/15 and 177 hours in 2015/16 (Average 200.5 hours/year) after implementation of the extended hours service.

Following initiation of the new pattern of working, the highest proportion of call outs were between 19:31 and 22.30. The large proportion of on-calls during this time period results purely from weekend days, where

the service moves to ‘on call’ at 20:00. Although this adds evidence to the requirement for a later finish on weekdays, extending the hours to 22:00 at a weekend would require two hours of extra paid overtime per day from the early shift team. This would result in a less cost efficient service, as this equates to more than the on-call cost during these hours. In addition, the early shift team were already being expected to work frequent weekends (although no on-calls), and longer shifts may add an additional burden to this team.

Introduction of the extended shifts, as expected, dramatically reduced overall call in rate, and this was especially seen in the first half of the shift. From around 03:30 onwards, the call-in rate was similar to the old on call pattern. In addition, staff were not expected to attend work until 13:45 after an on call, providing additional rest time in comparison to the previous expectation of 08:30.

1) Inappropriate call outs

Shortly after implementation of the extended pattern of working, it became evident that the increased presence on the ward was resulting in the service being seen less as emergency cover. The amount of call outs which had been recorded as ‘inappropriate’ (by the attending physiotherapist’s evaluation) on the electronic record were analysed and compared.

Prior to starting the new on call system, the inappropriate call out rate was 9.30%. Following one year of data collection, 16.75% of the out of hours on calls were recorded by the physiotherapist as inappropriate, demonstrating a large increase. Education was provided to the wards on the on-call service, however this did not reduce the inappropriate


Number of call outs per year

Increase / decrease compared to the preceding year

2011-12 205 0% change 826 calls in three years preceding new structure

2012-13 246 20% (Higher acuity patients on PICU)

2013-14 375 52% (Activity increasing on PICU)

2014-15 191 49% with new pattern of working 510 calls in the three years after the new structure 38.3% 2015-16 148 23%

2016-17 171 15%

TABLE 1: Call-out numbers, 2011 - 2017

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call out rate which has averaged 16.96% since implementation of the new pattern of working. Further education is required on this to ensure the service is being used appropriately and further reduce the inappropriate call in rate.

2) Orthopaedic Discharges

At the one year review, the weekend lists and weekday late book were used to identify orthopaedic patients who were discharged from physiotherapy following an intervention outside of normal working hours. Patients who were seen for mobility practice or exercises and still required on-going intervention (i.e. were not discharged following physiotherapy) were not included.

Over the year, 71 patients were seen and discharged on a weekday late shift and 63 orthopaedic patients

were seen and discharged on a weekend shift.

When both the weekday and weekend service was considered, 134 orthopaedic patients were discharged home following physiotherapy on an evening or a weekend. This reduces a minimum of 134 bed days per annum with the associated cost savings and improved patient and carer satisfaction. More bed days may be saved than this as previously some patients were staying in for the whole weekend to access physiotherapy on the Monday.

Financial Implications of the new structure

An extra member of staff (1WTE band 6) was funded by the Trust to enable the new pattern of working. As of April 2017, a mid-point band 6 would receive £29,301 per annum and cost the Trust £37,169 annually including all on costs. This was to be offset by an


FIGURE 3: Number of call outs per year

The time of call out was analysed for two complete years from the electronic record, and the average of these numbers was compared with average data for the previous three years. This is demonstrated in Figure 4.

FIGURE 4: Time of call outs

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assumed financial savings in on call hours and also weekend hours being paid as TOIL rather than paid overtime for late shifts. Table 2 demonstrates the cost of the ‘old’ and ‘new’ structures. As staff spanning grades 5-8a work on call, with the majority of staff being band 6, the mid-point band 6 hourly rate of £15.68 was used for calculations.

The financial calculations demonstrate that a higher rate of enhancements were paid in the new structure, but the cost of on call hours and weekend overtime was greatly reduced. The new pattern of working enabled patients to be discharged on evenings, presenting a large cost saving. This off set the financial outlay of the additional member of staff, leading in a reduction in cost of the on-call service from approximately £42,024 to £26,294.

Discussion

The results of the on-call service review have shown a clear, dramatic reduction in the number of overnight call-ins with introduction of the new structure. It also reduced the number of on calls prior to 03:30. Not only does this have a financial implication, but

it also represents more treatments being carried out on an evening in a controlled manner, rather than potentially allowing patients to deteriorate prior to an emergency call in.

In the previous structure, the nature of the small team resulted in frequent call-outs, which was becoming increasingly busy. Regular call outs occurred during the night resulting in the need for compensatory rest the following day. This may also impact on the reduction in staff sickness rates seen following the introduction of the new structure, where not only call in rates were reduced, but the times of call outs tended to be later. In addition, compensatory rest was easily enabled with the extra member of staff in the team, leading to more rested staff members.

Irrespective of financial considerations, the new structure allowed an increased quality of service. More patient treatments, for a wider variety of patients, were carried out in the new structure, on evenings and weekends. This enabled more flexibility for patients, who may have been in day surgery, or chronic respiratory patients who were more able to go out of the hospital in the afternoon and have an


TABLE 2: Approximate costs of the ‘old’ and ‘new’ structure

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evening treatment on their return. Additionally, this enabled more treatments to be carried out on evenings and weekends, when family members may be present to engage in the child’s therapy who may have been working during the day. Unfortunately, this increase in ‘visibility’ of the physiotherapy service late into evenings appears to have had an adverse impact on the rate of inappropriate call outs. This may also reflect the wider team respecting the physiotherapist’s opinion on a deteriorating child irrespective of whether this turns out to be a physiotherapy amenable issue.

For a change in service which is cost effective, this has also introduced an additional member of staff to the paediatric physiotherapy team. While this member of staff ‘backfills’ the staff member on a late shift, this person provides additional staffing in the afternoon if no compensatory rest is required. This enables additional flexibility within the team allowing improvements to the service delivered to patients, and service improvement projects.

Conclusions

Working an extended service within paediatric physiotherapy resulted in a large drop in call out rates, facilitation of orthopaedic discharges and no increase in staff sickness rates.

Financially, the establishment increased by one additional band 6 member of staff to enable adoption of this pattern of working. The overall cost of the on-call service reduced significantly when the associated cost savings were considered.

References

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Chartered Society of Physiotherapy. (April 2008). Seven day working, shift working and extended hours. London: CSP.

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Department of Health. (November 2012). The NHS Outcomes Framework 2013/14. London: HSMO.

Kuokkanen, L. et al. (2007). Organizational change and work-related empowerment. Journal of Nursing Management, 15, 500-507.

Mustain, J., Lowry, L. & Wilhoit, K. (2008). Change

readiness assessment for conversion to electronic medical records. The Journal of Nursing Administration, 38 (9), 379-385.

NHS. (January 2012). The Equality Delivery System for the NHS. London: COI for the NHS.

NHS Choices. (2011). NHS Choices, your health, your choices. Retrieved 17th March 2013, from, http://www.nhs.uk/choiceintheNHS/Yourchoices/Pages/Yourchoices.aspx

NHS Commissioning Board. (June 2012a). Securing Excellence in Commissioning Primary Care. London: NHS.

NHS Commissioning Board. (2013). Everyone Counts: Planning for Patients 2013/14. London: NHS.

NHS Improvement. (2011). Seven Day Working Case Studies. Retrieved 17th March 2013, from http://www.improvement.nhs.uk/7DayServices/SevenDayWorkingCaseStudies/tabid/219/Default.aspx

NHS Institute for Innovation and Improvement. (2013). Length of Stay – Reducing Length of Stay. Retrieved 17th March 2013, from http://www.institute.nhs.uk/quality_and_service_improvement_tools/

Nuffield Trust. (December 2012). The Funding Pressures Facing the NHS From 2010/11 to 2021/22: A Decade of Austerity? London: Nuffield Trust.

Rogers, E. (2002). Diffusion of preventive innovations. Addictive Behaviours, 27, 989-993.

Shanley, C. (2007). Management of change for nurses: Lessons from the discipline of organisational studies. Journal of Nursing Management, 15, 538-546.

Suc, J., Prokosch, H. & Ganslandt, T. (2009). Applicability of Lewin’s change management model in a hospital setting. Methods of Information in Medicine, 48, 419-428.

Tower Hamlets (February 2011). Performing for Excellence – our programme for improving quality at a lower cost. Retrieved 17th March 2013, from, http://www.TRUSTX.nhs.uk/for-clinicians/TRUST-X bulletin/news/news-archive/performing-for-excellence-our-programme-for-improving-quality-at-lower-cost

Equality Act 2010, c. 15. London: The Stationery Office.

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The Barriers and Facilitators to Implementing ‘The Daily Mile’™ in a UK Based Primary School: a Process Evaluation Protocol

Jennifer Harris [a*], Linda Jane Milnes [b], Gary Mountain [b]

[a] Physiotherapy Department, Doncaster & Bassetlaw Teaching Hospitals NHS Foundation Trust, Doncaster Royal Infirmary, Armthorpe Road, Doncaster, United Kingdom DN2 5LT

[b] School of Healthcare, Baines Wing, University of Leeds, Leeds, United Kingdom, LS2 9JT


Background and Purpose

As the physical activity rates of school children in the UK continue to remain significantly below recommended levels, interventions aiming to promote physical activity have gained increasing popularity. This article reports a protocol for a process evaluation of ‘The Daily Mile’™ in a UK based primary school.

Method

Using a multi-method process evaluation design developed in accordance with UK Medical Research Council and Health Research Authority guidelines, both quantitative and qualitative approaches will be used to examine implementation, mechanism of impact and contextual factors instrumental to the replication and/or wider implementation of this new school-based physical activity programme. The study will take place in a primary academy located in the UK implemented by 4 teachers and 2 teaching assistants to an anticipated purposive recruitment sample of 70 children ages 6-11 years. Split into two phases, phase one of the study will involve a 12-week trial of the intervention with concurrent quantitative data collection. A maximum variance sub-sample of 24-30 relevant stakeholders will be invited to participate in phase two focus groups. Specific steps are taken to address ethical issues relating to the assent of young children informed by previous school-based health research.

Conclusion

This protocol demonstrates the application of standardised definitions and components relating to evaluation of physical activity in schools and provides physiotherapists, public health professionals and decision-makers with information on how this health-enhancing physical activity intervention was tested for its feasibility and possible scale up with information to aid wider scale implementation.

Keywords: Schools, Physical activity, Children, Health promotion, Protocol, Process Evaluation

Introduction

Physical inactivity is well established as a major risk factor for a wide range of long-term health conditions and has been identified as the fourth leading risk factor for global mortality (Lee et al., 2012, Sallis et al., 2016). In the UK, physical inactivity and diet account for 14.3% of all ill-health, disability and early death (Murray et al., 2013) with an estimated cost to primary care trusts in England of £940 million and rising (British Heart Foundation, 2013).The World Health Organisation recommend that children and young people perform at least 60 minutes of moderate to vigorous physical activity every day (World Health Organization, 2010). However, 79% boys and 84% girls aged 5-15 years living in the UK fail to achieve this (Health and Social Care Information Centre, 2013). As a result, public health campaigns aim to develop successful schemes to promote physical activity in childhood and to develop long term and

sustainable plans to support all children to achieve 60 minutes of moderate to vigorous physical activity on a daily basis.

The determinants of physical activity in children and young people are complex and are influenced by factors at individual, social, environmental and policy levels (Dobbins et al., 2013, Bauman et al., 2012). A systematic review by Bauman et al (Bauman et al., 2012) suggest that self-efficacy (confidence of ability to be physically active in specific situations) and participation in school sports are positive determinants of physical activity in children and adolescents living in high income countries. Also at an individual level, the perception of ability to be physically active in certain situations is considered to be a determinant in older children but is inconclusive in younger children (Bauman et al., 2012). An association is noted with the availability of family support and participation in physical activity but none found for demographic or

J. Harris / APCP Journal Volume 9 Number 1 (2018)

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cultural factors (Bauman et al., 2012). Environmental factors such as walkability, traffic speed and volume, proximity of homes and key destinations (i.e. shops or school) and access to recreational facilities were all considered correlates of physical activity in childhood but this mostly affected activity in the leisure domain (Ding et al., 2011). Although tracking of physical activity levels through time requires further research (Bauman et al., 2012), some suggest that physical activity levels of children are mostly established by 11-12 years of age (Brodersen et al., 2007). As previous activity is also a determinant of childhood physical activity levels (Bauman et al., 2012), interventions proposing to influence physical activity across the lifespan (and before social and cultural factors start to influence) should target children of primary school age or below (Brodersen et al., 2007).

Although theories relating to genetic and evolutionary determinants are still in their infancy, interventions should also aim to include specific populations considered to be at higher risk that may not otherwise engage. Public health campaigns to increase childhood physical activity levels need, therefore, to consider psychological, interpersonal and environmental determinants when identifying mediators for new interventions and may require policies to support changes and ensure programmes are wide-reaching and inclusive (Bauman et al., 2012). Consideration of physical activity outside the domain of leisure-activity is also key.

Due to their influence over large cohorts of children and young people, schools offer a unique opportunity to promote health and well-being through physical activity (Public Health England, 2015, Heath et al., 2012). Physical activity programmes delivered within education settings have the potential to reach children from all genders, ethnicities, socio-economic, geographical and cultural backgrounds. Many primary schools in the UK have access to facilities with open spaces which may be limited in the wider community and teachers can play key roles as ‘moving professionals’ to inspire their pupils through education (Varney et al., 2014). Educational policies are strengthened by growing evidence in support of the association between academic achievement and physical activity levels of children and young people (Singh et al., 2012, Wittberg et al., 2012, Ofsted, 2014). Evidence suggests some schools have close links with the local community and offer the potential to inspire extra-curricular activities and encourage the wider community to share in the benefits (Public Health England, 2015).

In 2013, a review by Dobbins et al summarised evidence of the effectiveness of school-based interventions in promoting physical activity and fitness in children and adolescents (Dobbins et al., 2013). This review, updated from 2009, identified

randomised controlled trials from around the world assessing physical activity based health promotion and disease prevention interventions specifically implemented in the school setting. The review findings unequivocally support the use of school-based programmes to increase the rates and duration of physical activity and reduction in time spent watching television in younger (primary school-aged) children. It also signposts readers to factors that may influence treatment effect (for example encouraging more physical activity during the school day). The large variety of intervention types, outcomes under analysis, effect of contextual factors and cost-effectiveness do, however, limit the quality of the evidence and generalisability of the findings.

School-based physical activity programmes include a number of factors such as variability in outcomes, behaviours, target populations or flexibility in delivery that would suggest that they be considered complex interventions (Craig et al., 2008). Craig et al define complex interventions as those “that contain several interacting components” (Craig et al., 2008, p.7) whose efficacy and level of impact should not be considered without first establishing if it can work in practice (Bonell et al., 2012). In order for school-based physical activity programmes to achieve wide-scale population level health changes, an understanding of how physical activity interventions work in the ‘real world’ is essential (Reis et al., 2016). Process evaluations enhance data provided through outcome evaluations by allowing policy makers to further interpret findings and develop understanding of how they might apply in other contexts, or to other populations, ensuring long-term sustainability and cost-effectiveness (Moore et al., 2015b).

A systematic review of process evaluations of school-based physical activity programmes by Naylor et al (Naylor et al., 2015) established links between the level of implementation and health outcomes. However, the quality of the studies included limited the rigor of these findings. This may relate to limited data on important factors in the process evaluation of complex interventions such as fidelity, quality and responsiveness (Naylor et al., 2015, Moore et al., 2015b). Key themes influencing implementation were identified as time constraints, contextual appropriateness, delivery efficacy, supportive school climate and training and technical support but authors acknowledge limited feedback from other key stakeholders such as governors that may highlight community level factors not considered in the current body of evidence (Naylor et al., 2015). They also acknowledge the importance of linking data on dose (how much is delivered) and reach (how many of the target audience come into contact with the intervention) with other contextual factors within process evaluations and in evaluating interventions

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post scale-up as well as during efficacy trials or prior to replication to fully understand the various layers of implementation.

Launched in Scotland, ‘The Daily Mile’™ is a physical activity programme aiming to “to improve the physical, emotional and social health and wellbeing of our children” (The Daily Mile Foundation, 2017). The scheme encourages teachers to take pupils outdoors for 15 minutes each day to run or walk around. Well received by local communities, Education for Scotland, charities and the media, ‘The Daily Mile’ was acknowledged in the government’s new Childhood Obesity Strategy as a recommended initiative (The Daily Mile Foundation, 2017, HM Government, 2016). Early implementation has commenced in schools across the UK and beyond. By encouraging implementers to “keep it simple”, focusing on short but regular bursts of moderate to vigorous physical activity within the school curriculum, with no requirements for staff training, equipment, additional staffing or multiagency working, ‘The Daily Mile’ has the potential to be a sustainable and cost-effective technique to promote physical activity in schools (HM Government, 2016). There are no published process evaluation or feasibility studies at this time to support a successful wide-scale replication.

The Study

Aim and objectives

The aim of the proposed study is to consider the barriers and facilitators of implementation of ‘The Daily Mile’™ to a UK based primary school. Using standardised definitions and components recommended by MRC (Moore et al., 2014), our objectives are:

• To explore the response of students, parents, teaching staff and governors, following a twelve-

week implementation of ‘The Daily Mile’ programme and consider unexpected pathways or consequences, mediators and contextual factors affecting implementation.

• To determine what was delivered during implementation period through analysis of rates of fidelity, dose and reach of the intervention

• To identify the barriers and facilitators of sustainable implementation in preparation for further efficacy and outcome evaluation studies

Design

This will be a two phase study using a sequential transformative multiple method design guided by theory-based evaluation (Weiss, 1997) and realist evaluation (Pawson and Tilley, 1997). This design draws on guidance from the UK Medical Research Council ‘Process evaluation of complex interventions’ (Moore et al., 2014) and Health Research Authority protocol guidance (Health Research Authority, 2015). It is well-accepted that complex interventions will vary in their effectiveness depending on the manner in which the theories interact with their context (Bonell et al., 2012). A pragmatic paradigm approach is used in this study to reflect the multi-disciplinary approach and multi-methods required to address the aims and objectives set (Shannon-Baker, 2016).

Recruitment and consent

Pupils will be informed of the study using a colourful illustrated ‘storybook’ developed specifically for this research in collaboration with a children’s public involvement group. This ensures the aims and objectives, risks and benefits of participation are communicated in a developmentally appropriate manner in line with best practice guidance (NIHR Medicines for Children Research Network, 2014). Although efforts are planned to ensure ongoing


Inclusion criteria Teaching staff and pupils from at least one key stage one (KS1) class and one key

stage two (KS2) class attending an identified Primary Academy are eligible for

phase 1 and 2 of the study. The school principal and a sample of school governors

will also participate in phase 2.

Exclusion criteria Any person advised against participating by a doctor or qualified health profession-

al will be excluded from phase 1 of the study. Consent / assent of all participants

will be reviewed before, during and after all discussions.

Table 1: Inclusion and exclusion criteria

Sample

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assent, pupil participants will all be under 18 years of age. Parents will, therefore, be asked to consent to participate on behalf of their child after reviewing parent/carer information leaflets distributed by teachers. Parents consenting on behalf of phase one participants will do so via an ‘opt-out’ process to reflect the ’whole-school’ approach advocated by ‘The Daily Mile’. This process is commonly applied in studies requiring high rates of participants such as school-based studies and is considered valid when both the child and parents are aware of all options (Milnes and Kendal, 2012, Spence et al., 2015). Awareness of all options for consent will be addressed via written information leaflets, open meetings at school and playground ‘meet and greet’ where parents and pupils will have the opportunity to discuss queries or concerns with the research team. Text message reminders will also facilitate return of ‘opt-out’ documents. Separate information sheets will also be provided to, teaching staff, school principal, school governors and phase two pupil and parent participants. ‘Opt-in’ consent forms will be used by all phase two participants due to burden of inconvenience and potential emotional impact associated with focus group participation and lower requirement for participation rates.

Phase 1

Participants and setting

A purposive sample of 40 key stage one pupils and 30 key stage two pupils will be taken from one primary school based in the East Midlands. They will be supported by 4 teachers, and 2 teaching assistants reflecting national local authority maintained primary school ratios (Economics Evaluation and Appraisal Team, 2011). Set in a rural region, the school lies in a lower level super output area ranked 13,906 out of possible 32,844 for indices of deprivation affecting children (where 1 is the most deprived) and average levels of free school meals (Opendatacommunities.org, 2015). The school is a slightly smaller than average sponsor-led academy catering for 272 children aged 3-11 years.

It is acknowledged that the development of ‘The Daily Mile’ intervention was driven by professional teaching experience, common sense and awareness of current health and well-being policies rather than existing theories. However, it is assumed that behaviour change models are likely to have underpinned some of the design considerations of this particular intervention. The theory of planned behaviour model (Ajzen, 1985) describes three components of behaviour (attitude, subjective norms and perceived behavioural control) all of which have potential to be influenced by The Daily Mile intervention and a logic model demonstrating these causal assumptions is detailed within Appendix 1. Although such models are well-applied in health behaviour programmes, their short-

comings are acknowledged (Barley and Lawson, 2016) and new insights are required that capture the complexities involved when individuals make life-changing decisions if innovative models are to be produced. As recommended by Moore et al. (2015a), local implementers will be supplied with established training resources developed by The Daily Mile Foundation which are publicly available on ‘The Daily Mile’ website (The Daily Mile Foundation, 2017). This reflects central implementation of the intervention. To avoid loss of credibility, the evaluators will retain a passive role throughout the study encouraging intervention queries to be referred to developers and not informing implementers of emerging findings.

Purposely designed ‘daily physical activity record sheets’ will be completed by teachers during the implementation period describing the number of occasions that each pupil participates in ‘The Daily Mile’ and how many complete the full 15 minutes. Implementers will also ask pupils to complete the p children’s OMNI perceived exertion scale at key points throughout the implementation period. This has demonstrated good concurrent validity with VO2 at 6-8 yrs (0.67) and 9-10 yrs (0.77) and reliability coefficient ranging 0.91 to 0.95 (Rice et al., 2010). Implementers will present each pupil with the scale immediately following ‘The Daily Mile’ at the close of weeks 1, 4, 8 and 12. Structured observation of the intervention will triangulate quantitative findings relating to fidelity and reach and qualitative data relating to mechanism of impact. Observation will follow a pre-determined schedule adapted from the SOFIT (System for Observing Fitness Instruction Time). Designed to simultaneously measure physical activity levels during physical education, class content and teacher behaviour in context, SOFIT has demonstrated good internal consistency reliability with coefficients 0.98 – 0.99 (Rowe et al., 1997), and is well-validated for face, construct and concurrent validity with heart rate and energy expenditure (Chan and Hui, 2011, McKenzie et al., 1992, McKenzie, 1991). Modification of phase 2 permits additional data on student behaviour and 9 observation periods per pupil (3 compared to 4 minutes) cater for the shorter timescale of ‘The Daily Mile’ compared to average PE sessions. Informal piloting of modifications will take place using SOFIT training videos.

Data analysis

Quantitative data will be summarised using descriptive statistics to describe measures of central tendency and variability (such as frequency, proportions range, means and standard deviation). Perceived exertion will be presented as medians and interquartile ranges. Summary scores from SOFIT physical activity expenditure will report the minutes and percentage of total intervention period from which percentage


41

MVPA, energy expenditure rate and total energy expenditure can be estimated. Context variables will be reported in terms of percentage of intervals. Data will also be summarised in graphical form comparing the percentage of MVPA with differing teacher behaviour categories and pupil behaviour categories.

Phase 2

Participants and setting

A purposive maximum variance sub-sample of 4-5 pupils from each age category will be selected by the primary investigator during phase 1 observation. These pupils will be invited to attend developmentally appropriate focus group discussions in the school setting. A parent or carer of each child selected will also be invited to attend a separate focus group. A final focus group will involve the implementers (teachers)

and other key stakeholders (school principal and governors).

Data collection

Four focus groups will be conducted with;

1. 4-5 key stage 1 pupils

2. 4-5 key stage 2 pupils

3. 8-10 parents/carers

4. 8-10 teaching staff, head-teachers and governors

Focus groups with the children will offer an ‘insider’ perspective of the intervention including mediators such as attitudes towards physical activity. Focus groups will follow an age and developmentally appropriate structure and plan acknowledging lessons learnt from previous focus groups in school-


Objective Description of methods Data source Time point

To determine what was delivered during implementation period through analysis of rates of fidelity, dose and reach of the intervention

SOFIT structured observation KS1 pupils

KS2 pupils

Teaching staff (im-

plementers)

Phase 1

(Sep - Dec)

Focus group discussions KS1 pupilsKS2 pupilsParents/carersTeaching staff and governors

Phase 2(Feb - Mar)

To identify the barriers and facilitators of sustainable implementation in preparation for further efficacy and outcome evaluation studies

Daily physical activity record sheets

Teaching staff (implementers)

Phase 1(Sep - Dec)

OMNI children’s perceived exertion scale

KS1 pupilsKS2 pupils

Phase 1(Sep - Dec)

SOFIT structured observation KS1 pupilsKS2 pupilsTeaching staff (implementers)

Phase 1(Sep - Dec)

To identify the barriers and facilitators of sustainable implementation in preparation for further efficacy and outcome evaluation studies

Focus group discussions KS1 pupilsKS2 pupilsParents/carersTeaching staff and governors

Phase 2(Feb - Mar)

Table 2: summary of outcome measure

Data collection

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settings (Spyrou, 2011). The ‘draw and talk’ technique will be applied in both focus groups with children to allow younger children or those with poorer language development to respond to a question or prompt with a drawing and then elaborate the meaning of the picture orally (Mayaba and Wood, 2015). Pre-determined topic guides using iterative data from phase one will include key themes, cues and prompts and encourage peer interaction and facilitate spontaneity. Adult focus groups will provide an ‘outsider’ view on the intervention offering more detail on how the intervention affects teaching systems and organisations and any wider effects on a social or community level. Topic guides and prompts will be applied once more, informed by phase 1 quantitative data collected in an inductive process and by themes highlighted by Naylor et al. (2015) in their implementation review. All focus groups will be facilitated by one moderator, audio-recorded and transcribed verbatim. Reflective diary will be maintained throughout data collection and analysis and pre-conceptions recorded to facilitate reflexivity.

Data analysis

Basic (or descriptive) quantitative measures of implementation will be combined with in-depth qualitative data to provide detailed understandings of the intervention functioning on a small scale. Focus group transcripts will be categorised and coded thematically based on the framework method described by Gale et al (Gale et al., 2013). Quantitative and qualitative data will be analysed separately but with consideration of the merits of each data type and triangulation of the results discussed within the findings. Credibility of data will be established through various means (Silverman, 2011). Summaries of main topics will be discussed after focus groups to offer opportunity for respondent validation. Co-authors will operate as independent reviewers for data comparison and coding structure and will seek out disconfirming or inconsistent information. Declaration of preconceptions and beliefs will be recorded prior to study and a reflexive journal will be taken throughout the data collection and analysis period to maintain awareness of how these assumptions shape or influence both analysis and data collection.

Data management

Pupils will be allocated a unique identification number by implementers and evaluators will not have access to participant-identifiable information. All data will be stored on a secure server that requires encrypted access in accordance with Data Protection Act (1998). Only the research team will have access to the data. All reasonable efforts will be taken to maintain confidentiality and anonymity during focus group discussions.

Ethical considerations

This research evaluation has received ethical approval the University of Leeds, School of Healthcare Research Ethics Committee (SHREC) (date of approval 27th May 2016 ref:HREC15-055) A formal amendment is required for any modification of the protocol and needs approval by the SHREC..

Discussion

The study protocol presented here details how ‘The Daily Mile’ will be process evaluated using a combination of quantitative and qualitative measures. The study incorporates the recommended key dimensions of a robust process evaluation by examining implementation, mechanism of impact and contextual factors (Moore et al., 2014) and builds on evidence from previous process evaluations and reviews of school-based physical activity programmes by focusing on physical activity implemented within the school day and applying standardised definitions and measures to evaluation (Dobbins et al., 2013, Naylor et al., 2015). This includes validated outcome measures selected to reflect the age and development of participants and the environment in which the intervention is applied. Methods triangulation will ensure that the evaluation findings are rich, robust, comprehensive and well-developed (Patton, 1999). Data collection at varying time intervals reflect changes in implementation, behaviour and physical fitness and the passive role of the evaluator should reduce changes as a result of measurement and observation.

It is predicted that the outcomes that will emerge will add to existing knowledge related to school-based physical activity and provide vital information to decision makers and implementers regarding refinement of logic models to consider impact and how the intervention might be replicated and/or scaled up. This will aid the national strategy to develop an acceptable, sustainable and effective programme to increase the physical activity levels of a wide-reaching and diverse population of children aged 5-11 years as recommended by NICE (NICE, 2009).

Ethical Approval: University of Leeds, School of Healthcare Research Ethics Committee (SHREC) (date of approval 27th May 2016 ref:HREC15-055).

Funding: This report is independent research supported by the Health Education England / National Institute for Health Research Integrated Clinical Academic (ICA) Programme Masters in Clinical Research Studentship Scheme MRES-2015-03-020. The views expressed in this publication are those of the author(s) and not necessarily those of the NHS, the National Institute for Health Research or the


43

Department of Health. An Association of Paediatric Chartered Physiotherapists Research Bursary supported costs relating to participant information booklet illustrations and printing and data collection boxes.

Conflict of Interest: There are no conflicts of interest

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Gemma Kelly BSC (Hons) Physiotherapy, MSc Neurophysiotherapy [*] Jackie Shanley PgDip Physiotherapy, MSc Neurorehabilitation, PgCert Learning and Teaching in Higher

Education


Introduction

Physiotherapists are required to make clinical decisions by integrating the best available research evidence with clinical expertise and child and family preference (CSP, 2014). This evidence based practice promotes the use of interventions that offer the best outcomes for the child and family, and are cost effective (Moitra and Neogi, 2016). However, a lack of studies investigating some paediatric client groups and interventions offered by physiotherapists can make engaging in evidence based practice challenging. With increasing demands for physiotherapists to use research evidence to justify the services they provide (Skinner et al. 2015), there is a need to promote and develop all forms of research with this diverse and complex client group.

Single case experimental design (SCED) is a form of quantitative methodology that has the potential to produce highly relevant paediatric physiotherapy research. In this type of design, the subject’s performance in normal circumstances is compared to their performance in experimental circumstances (e.g. with the addition of an intervention) (Portney and Watkins, 2014). This form of research has traditionally been overlooked in favour of group based designs, which are perceived to produce higher quality research. However, with the recent developments of quality standards for SCED research, Evans et al. (2014) argue that SCED is a rigorous research methodology that can produce results that are useful and meaningful in rehabilitation.

This article will explore SCED and its place within the quantitative research paradigm, and subsequently evaluate its use within paediatric physiotherapy research. Finally, it will consider how paediatric physiotherapists may utilise SCED as a method to carry out relevant research within their own clinical environment.

Discussion

SCED is a quantitative research methodology, and it therefore adopts the philosophy of the positive research domain by assuming that there is one reality, and that accurate and detailed information can be gained about this reality (Bowling, 2014). The research question is answered through systematic collection of data by observation and measurement (Rolfe et al,

2013). As a form of quantitative methodology, SCED can be used to address clinical questions that require a yes or no answer, and is most commonly utilised to determine whether an intervention is better than routine or no treatment (Kratchowill et al. 2010). An example of a research question that could be targeted with SCED methodology is: Does the addition of functional electrical stimulation (FES) to standard rehabilitation improve the upper limb function of a child with an acquired brain injury? SCED differs from case study methodology, which involves the retrospective, in-depth exploration of a child or group of children in their natural, real life environment. (Crowe et al., 2011). In comparison, SCED requires predetermined research protocols and the systematic collection of repeated measures of the effect of the intervention (Smith, 2012). Additionally, SCED, being a form of research, will require ethical approval from the appropriate ethical governance committee (for example Health Research Authority approval if the research is being conducted within the NHS) prior to the research being conducted. This will ensure the research is conducted appropriately and the child and family’s physical and emotional well-being are protected (Bell, 2008).

The underlying philosophy of the SCED approach is the same as group based experimental methods, such as randomised controlled trials (RCTs). However, the methodology is different. In an RCT, participants are allocated into groups, and one group is offered the intervention and the other group acts as a control (with either no intervention or routine practice). The target behaviour (e.g. upper limb function) is measured before and after the intervention and any differences in behaviour between the two groups are analysed (Portney and Watkins, 2014). SCED studies investigate the effect of the intervention (for example, FES), on one individual participant. This is achieved through taking regular measurements of the target behaviour (e.g. upper limb function) over a period of time: during a baseline (or control) phase and then during an intervention phase where the intervention of interest is introduced (for example FES). The series of measurements (data points) in the intervention phase are then compared to those in the baseline phase (Portney and Watkins, 2014; Smith, 2012). Any differences between the phases would suggest that the intervention produced a treatment effect in the subject.

G. Kelly / APCP Journal Volume 9 Number 1 (2018)

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A further difference between SCED and RCTs is the way in which the data is analysed. RCTs rely on the descriptive analysis of the difference in outcomes between the different groups, with statistical testing to determine whether differences seen are significant or due to chance. In SCED the data are analysed through the comparison of the level (mean of the data scores for that phase), and trend (direction and gradient of change within a phase) between baseline and intervention phases (see figure 1) (Tate and Perdices, 2015). The direction of change is observed from the trendline, and categorised as either accelerating, decelerating or staying stable. The gradient of the trendline can also be analysed to indicate the rate of change (Portney and Watkins, 2014). Statistical analysis can be used in addition to visual analysis, to indicate the significance of any changes between adjacent phases (Kratochwill et al. 2010). The difficulty in using statistical testing to analyse SCED data is that the data are likely to be serially dependent, meaning that each data point is related to the next. One of the pre-requisites of statistical testing is that data are independent of each other. Thus it is important that SCED data are tested for serial dependency by using the Durbin-Watson statistic (Portney and Watkins, 2014). If serial dependency is not present statistical testing such as the C-Statistic (Portney and Watkins, 2014) can be used. If serial dependency is present, visual analysis of the data can be augmented by a semi-statistical analysis, for example by using the two standard deviation band method, which can indicate whether changes seen with the visual analysis are

significant (Portney and Watkins, 2014). Therefore, SCED data needs to be analysed visually, tested for serial dependency, and the most appropriate statistical or semi-statistical technique selected to test for significance of any changes found.

A minimum of five data points per phase is recommended to enable visual analysis of SCED data (Smith, 2012). One of the key requirements of SCED is the establishment of a stable baseline, and therefore the baseline phase may need to be extended above the five data points to allow stability to be established (Kratochwill et al. 2010). However achieving stability in the baseline phase can be challenging or impossible when conducting physiotherapy research with some children. Children often vary in their performance of skills, and for some groups of children, a baseline that has a continuous trend would be expected. For example, children in the subacute stage following acquired brain injury may not reach stability in their skill performance for a long time, as they may still be experiencing natural recovery. However, given that it is important to establish which intervention (if any) maximises their recovery of skills and function, simply excluding this group of children from research is not in their best interests. Instead, using multiple intervention and withdrawal phases, or use of multiple baseline designs as discussed below, could be used to ensure that any effects of the intervention that are seen are replicated across intervention phases or subjects, and thus increasing the validity of the study.


Figure 1: Example of a visual analysis graph of SCED data.

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Withdrawal designs

There are several different variations of SCED (Rassanfiani and Sahaf, 2010; Smith 2012, Kratchowill et al. 2010). The first, known as the withdrawal design, or ABA design, is where there is a baseline phase, followed by an intervention phase, followed by a phase where the intervention is withdrawn. Thus, the researcher can observe whether changes noted in the intervention phase continue after withdrawal of intervention (natural history of behaviour) or whether the changes plateau or reverse when the intervention is withdrawn (suggesting that any change observed in the intervention change was due to the intervention) (Rassanfiani and Sahaf, 2010). An ABAB design, where an additional intervention phase is added after the withdrawal phase, will further increase confidence that any changes seen are due to the intervention and not to chance (Portney and Watkins, 2014). Therefore adding a further cycle of non-intervention and intervention phases can increase the internal validity of the study.

Multiple baseline designs

A second variation of SCED research is the multiple baseline design, where SCED methodology is used on multiple individuals (Rassanfiani and Sahaf, 2010). If similar changes are seen in the target behaviour of three or more individuals with similar characteristics, there is increased confidence that the results seen are due to the intervention (Portney and Watkins, 2014). Varying the length of the baseline will further ensure that any results seen are not due to chance (Smith, 2012). Withdrawal designs (e.g. ABA, or ABAB designs) could be used with multiple individuals to further increase the reliability and validity of results (Portney and Watkins, 2014). As well as increasing the internal validity of the study, completing the research on multiple individuals can also increases the external validity of the study, as it would suggest that the intervention had (or did not have) an effect on multiple individuals (Smith, 2012).

Alternating treatment designs

The third variety of SCED methodology is alternating treatment designs (ABC designs) where the researcher changes two or more interventions over the timeline (Rassanfiani and Sahaf, 2010). This will allow the comparison of the effects of the different interventions on the individual.

Benefits of SCED methodology in paediatric physiotherapy research

Paediatric physiotherapists work with many rare or heterogeneous populations, and therefore recruiting enough participants with similar characteristics to power a group based experimental research

design can be challenging (Tate and Perdices, 2015). SCED can overcome this challenge by providing a method for rigorously investigating physiotherapy interventions with these children (Graham et al. 2012). This can allow the development of an evidence base for children for whom very little evidence currently exists. Additionally, RCTs are often intensive of time and resources (Portney and Watkins, 2014), which makes undertaking this form of research very challenging for clinical physiotherapists. Although SCED research requires investments of time to establish protocols, gain ethical approval, collect and analyse the data, the use of SCED is much more feasible to be carried out alongside clinical practice.

Group based research designs allow the generation of evidence regarding the average response of a group of children to standardised interventions. A criticism of this is that this is not reflective of clinical practice (Tincani and Travers, 2017), as therapy is catered to the needs of individual children, and different children respond differently to the intervention. In SCED, treatment methods can be much more reflective of true clinical practice, and it allows an understanding of the effects of the intervention on the individual, which provides clinically rich and meaningful data (Vannest and Ninel, 2015). Furthermore, SCED allows the effects of the intervention on the individual to be studied over time, which can provide useful information about the nature of recovery of skills or function with rehabilitation (Graham et al. 2012). For example, it is known that when learning a motor skill, performance can be variable, and can be influenced by factors other than the child’s motor ability (Schmidt and Lee 2005). This variability in performance can be captured in SCED research, and can be useful to allow therapists to understand the trajectory their patients may follow with an intervention (Rassafini and Sahaf 2010). This not only provides useful clinical information, but is also important in determining the true impact of the intervention, as measuring at one point in time may not have captured the child’s true abilities, and given either falsely negative or falsely positive results.

Limitations of SCED methodology in paediatric physiotherapy research

Although benefits of using SCED methodology have been highlighted, there are also limitations to its use which need to be considered (Table 1). One of the main limitations to SCED research is that its external validity is limited due to the fact that it studies an individual case. A well conducted SCED study could only provide evidence that the intervention was successful or unsuccessful for the individual who was studied at that point in time. Nonetheless, the process of rigorously determining the effect of the intervention in one individual will generate knowledge which can be considered by therapists in their clinical practice


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and be used as part of their clinical reasoning when considering other, similar cases (Vannest and Ninel, 2015). Furthermore if multiple studies are conducted with children with similar characteristics, or on similar interventions, the knowledge base can further grow (Evan et al. 2014), and an understanding of how different children react to the intervention can be reliably gained.

A second limitation that could occur in SCED research is the risk of bias. SCED research involves continuous measurement for the period of the study. Ideally, the data collector should be blinded to the phase of the study (Smith, 2012), however, in reality this can be challenging when data collection is occurring within a physiotherapy department multiple times a week. The use of strict measurement protocols, and if possible, selecting assessments which are scored via filming, so the therapist completing the scoring could be blinded as to the phase of the study, may help reduce the risk of bias. As in all research, steps must also be taken to ensure that the assessments are chosen are reliable and sensitive to change, and that the intra and inter-rater reliability of the assessments are high (Portney and Watkins, 2014). Furthermore, the outcome measures need to be selected carefully, as the child will undergo repeated assessments. Therefore, assessments that are long and burdensome should be avoided. Thus minimising bias, and selection of appropriate outcome measures must be an important parts of planning a SCED study.

A third limitation to SCED is the ethical concerns associated with a phase of withholding the intervention. There will be a baseline phase where the intervention is not offered, and potentially a withdrawal phase where the intervention is stopped for a period of time (Portney and Watkins, 2014). Consideration will need to be given as to any detrimental effects these non-intervention phases may have on the child. There are practical solutions to overcoming this concern, such as using waiting times or periods of non-contact for the baseline phase. Alternatively, maintaining normal treatment as the baseline, and adding the intervention to normal treatment during the intervention phase could reduce

the ethical concerns of withholding a treatment.

Does SCED research have a place in paediatric physiotherapy research?

From the discussion, it is clear that despite limitations to SCED research, there is a substantial place for its use in developing the evidence base for paediatric physiotherapy. As with all forms of research, the quality of each SCED study needs to be critically evaluated before utilising the findings in clinical practice. There are now several checklists that can be used to help establish the reliability and validity of SCED research, and ensure it contains sufficient information about the subject and intervention to allow it’s use (Evans et al., 2014). It is likely that one piece of SCED research alone will not provide sufficient evidence to justify an intervention for a certain population. Yet if the knowledge gained from the response of a child with specified characteristics is combined with other sources of evidence, such as other research studies, clinical expertise, or patient preference it can assist the therapist’s clinical reasoning process. SCED can also lead to the development of group based research studies, or further SCED research on similar topics (Graham et al. 2012), and the results can be combined and further increase the reliability and validity of the research.

Conducting SCED research

Physiotherapists are required to make many clinical decisions within their day to day practice, which ideally would be supported by relevant research (Scurlock-Evans et al. 2014). SCED is one type of research methodology which paediatric physiotherapists could conduct within their working environment to develop the evidence base for the clinical decisions they need to make. There is no doubt that conducting a piece of research requires time and resources, even if the intervention that is being studied is part of what is offered in routine clinical practice. However formally and systematically investigating clinical questions to provide research evidence can bring many benefits.

Completing the research will require time and effort commitments from the researcher, their team and


Benefits of SCED research Challenges of SCED research

• Can be used with heterogeneous or low incidence populations

• Feasible to be conducted alongside clinical practice

• Allows investigation of an intervention in a realistic clinical environment

• Gives detailed information about the response of the individual over time

• Limited external validity • Risk of bias• Assessment burden to the child • Ethical concerns of a baseline phase (and

potentially a withdrawal phase) where the intervention is not offered

• Challenge of establishing a stable baseline for some groups of children

Table 1: Summary of benefits and challenges of SCED

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the child and family. The child will have far more assessments than is routine in clinical practice, and the team working with the child may need to be involved in data collection. However, if assessments are short, relevant and enjoyable for the child, the burden is minimised. The researcher will require time and resources to develop protocols, gain ethical approval, complete assessments, analyse data and write and disseminate findings. Although this can be daunting, as SCED is investigating interventions with an individual within a real clinical environment the process can be relatively straight forward, and there are many texts that can guide the researcher through the process (for example, Portney and Watkins, 2014).

Overall, although there will be challenges associated with conducting research, the benefits of developing the evidence base for paediatric physiotherapy to ensure that it is efficient and cost effective are undisputable (Skinner et al. 2014). Furthermore there are multiple benefits to the physiotherapist conducting the research that can enhance their clinical practice, and the service in which they work. These can include professional development for the individual and their team, and increased job satisfaction from the challenges and rewards of participating in research (Wenke et al. 2017). Finally, it has been shown that having research activity in the department can bring benefits at an organisational level. Departments that have an active research culture are likely to have higher levels of patient care and better retention of their staff (Harding et al. 2017). Therefore, there are many benefits that can compensate for the challenges and effort that are required to undertake SCED research.

Conclusion

SCED is a robust form of quantitative research methodology that explores the effects of an intervention on an individual. Despite some limitations it offers unique benefits that promote its inclusion in paediatric physiotherapy research. Conducting the research will bring challenges to physiotherapists, but also benefits to the physiotherapist completing the research, the department they work in, and the children and families they work with. Furthermore, developing the evidence base for paediatric physiotherapy will benefit the profession by ensuring that the most effective and efficient interventions are offered to all children and families.

References

Bell, N., 2008. Ethics in child research: rights, reason and responsibilities. Children's Geographies, 6(1), pp.7-20.

Bowling, A. (2014) Research Methods in Health: Investigating Health and Health Services. 4th edn.

Berkshire: McGraw-Hill Education

Crowe, S., Cresswell, K., Robertson, A., Huby, G., Avery, A., & Sheikh, A. (2011). The case study approach. BMC medical research methodology, 11(1), 100.

CSP (2014) 'What is evidence based practice?' Available at http://www.csp.org.uk/professional-union/practice/evidence-base/evidence-based-practice/what-evidence-based-practice [2.1.2018]

Evans, J. J., Gast, D. L., Perdices, M., & Manolov, R. (2014). Single case experimental designs: Introduction to a special issue of Neuropsychological Rehabilitation. Neuropsychological rehabilitation, 24(3-4), 305-314.

Graham, J., Karmarkar, A., and Ottenbacher, K. (2012) 'Small Sample Research Designs for Evidence-Based Rehabilitation: Issues and Methods'. Archives of Physical Medicine and Rehabilitation 93 (8), S111-S116

Harding, K., Lynch, L., Porter, J. and Taylor, N.F., 2017. Organisational benefits of a strong research culture in a health service: a systematic review. Australian Health Review, 41(1), pp.45-53.

Kratochwill, T., Hitchcock, J., Horner, R., Levin, J., Odom, S.. Rindskopf, D., Shadish, W.. (2010) What works clearinghouse: single case design technical documentation, Available from http://files.eric.ed.gov/fulltext/ED510743.pdf, [2.1.2018]

Moitra, M. and Neogi, M., 2016. Evidence-based Physiotherapy-Self reported attitude and belief among physiotherapists: A cross-sectional study. Integrated Research Advances, 3(2), pp.45-46.

Portney and Watkins (2014) Foundations of clinical research: applications to practice: 3rd Edition, Edinburgh; Pearson Education Limited

Rassafiani, M. and Sahaf, R. (2010) 'Single Case Experimental Design: An Overview'. International Journal of Therapy and Rehabilitation 17 (6), 285-289

Rolfe, G. (2013) ‘Philosophical basis for research’ in Curtis, E. and Drennan J., Quantitative Health Research: Issues and Methods’ London: McGraw-Hill Education, 11-29

Schmidt and Lee (2005) Motor control and learning, 4th edition, Leeds; Human Kinetics

Scurlock-Evans, L., Upton, P., & Upton, D. (2014). Evidence-based practice in physiotherapy: a systematic review of barriers, enablers and interventions. Physiotherapy, 100(3), 208-219.

Smith, J. D. (2012) 'Single-Case Experimental Designs: A Systematic Review of Published Research and Current Standards.'. Psychological Methods 17 (4), 510

Skinner, E.H., Williams, C.M. and Haines, T.P., 2015.


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Embedding research culture and productivity in hospital physiotherapy departments: challenges and opportunities. Australian Health Review, 39(3), pp.312-314.

Tate, R.L. and Perdices, M.A. (2015) ‘Single case experimental designs’ in Hammond F.M., Malec, J.F., Nick, T.G. and Bushbacher R.M. Handbook for clinical research; Design, statistics and implementation, DemosMedical: New York, pg 13-18

Tincani, M. and Travers, J., 2017. Publishing single-case research design studies that do not demonstrate experimental control. Remedial and Special Education, p.0741932517697447.

Vannest, K. J. and Ninci, J. (2015) 'Evaluating Intervention Effects in Single-Case Research Designs'. Journal of Counseling & Development 93 (4), 403-411

Wenke, R.J., Ward, E.C., Hickman, I., Hulcombe, J., Phillips, R. and Mickan, S., 2017. Allied health research positions: a qualitative evaluation of their impact. Health research policy and systems, 15(1), p.6.


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Virginia Knox MRes, MSC, PGCE, MCSP The Bobath Centre, London


Introduction

The Pediatric Evaluation of Disability Inventory-Computer Adaptive Test (PEDI-CAT) (H. Dumas et al., 2010; Haley et al., 2012) measures typical performance of functional skills in children and youth aged up to 21 years, with a range of physical, cognitive and/or behavioural conditions. It can be completed by a parent or a health professional familiar with the child. A computer adaptive format permits statistical modelling to calculate a valid score and ensures that the minimum number of items are tested (Haley et al., 2011).

The PEDI-CAT measures functional skills in four domains: Daily Activities, Mobility, Social/Cognitive, and Responsibility. Each domain is divided into content areas containing individual items. Within the PEDI-CAT’s Mobility domain, there is a 12-item manual wheelchair sub-domain intended for children who self-propel their wheelchairs (H. Dumas et al., 2010). Currently, the PEDI-CAT Wheelchair subdomain does not have any power wheelchair items and very few items on the easier end of the wheelchair scale applicable for children who do not self-propel a manual chair.

Response options for the first three functional domains are on a 4-point scale: from ‘unable’, ‘hard’, ‘a little hard’ to ‘easy’. Response options for the fourth domain (Responsibility) range from the adult having full responsibility to the child having full responsibility on a 5-point scale. The PEDI-CAT generates both a normative score (T-score and age percentile) as well as a scaled score. The PEDI-CAT’s scaled scoring was developed with a 20-80 scaling metric with the intent that new items could be added at the extremes of the score ranges (0-20 and 80-100) (H. Dumas et al., 2010).

A specific need for additional wheelchair items for the PEDI-CAT’s Mobility domain has been identified in recent validation studies (Dumas & Fragala-Pinkham, 2012; Fragala-Pinkham et al., 2016). Modelling previous item development procedures used for the PEDI-CAT (H Dumas et al., 2010; H. Dumas et al., 2010) the purpose of this study was to develop a potentially new item pool The purpose of this study was to develop potentially new items for the PEDI-CAT Wheelchair subdomain for children who self-propel manual wheelchairs, drive powered chairs or are pushed in a wheelchair. This was part of a larger

study to identify areas of the PEDI-CAT functional mobility continuum that could be enhanced with additional items.

Method

Participants

Purposive convenience samples of physical therapists and physiotherapists (PTs) were recruited for three focus groups, from an acute paediatric rehabilitation hospital in the USA (n=6; female=6, male =0), a paediatric outpatient treatment clinic in the UK (n=3; male = 1, female = 2) and from a UK paediatric community team (included home visiting, under-five provision, special and mainstream schools) (n=8; male=1; female =7). Inclusion criteria were that the paediatric therapists needed experience working with children with limitations in mobility and/or who self-propelled manual wheelchairs or drove power chairs. Therapists were recruited in the USA and UK to try to ensure the content of the items would be valid to children in both countries.

Therapists from these focus groups were later recruited via e-mail or in-person to participate in further item refinement through cognitive interviews(Dumas et al., 2008), an interview strategy used to gain insights into how potential respondents understand and respond to questionnaire items. Written informed consent was obtained prior to participation in the focus group and/or cognitive interview indicating that the participant’s identity will be known to the investigator(s) and other participating therapists in the focus group (as data is collected in an open forum). Those who choose to participate in a follow-up cognitive interview or participate in the on-line survey were not identified other than to this study’s investigators.

Therapists provided demographic information including: number of years working as a physiotherapist, place of work, (e.g. early intervention, special schools) and description of children on their caseload (e.g. age range, GMFCS levels). No monetary honorariums were provided but as the groups took place during lunchtimes, refreshments were provided. This study was subject to full ethical review and approval was given by the Institutional Review Board at Franciscan Hospital for Children, Boston, MA, USA and by the National Research Ethics Service via the Royal Free Hospital Research and Development Department.

V. Knox / APCP Journal Volume 9 Number 1 (2018)

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Procedure

The procedures for this item pool development study included a literature review, focus groups and cognitive interviews (Artino et al., 2014; H. Dumas et al., 2010; Walsh et al., 2008).

First, a literature review searching for outcome measures for manual and power wheelchair skills that might be appropriate for all levels and type of wheelchair user, using search engines including CINAHL, Medline, PubMed and Cochrane reviews was compiled. The items from each of the measures were reviewed by the investigators and the content was used to guide question development for focus groups of physical therapists and item development.

Next, three focus groups were led by 1-2 of this study’s investigators who are familiar with the PEDI-CAT. The purpose of the focus groups was to gather thoughts and ideas about item content to enhance the PEDI-CAT item pool. Each focus group lasted 60-90 minutes. Focus group questions for the discussion of wheelchair skills, included:

1. Do you use a specific measure for evaluating wheelchair skills in children?

2. What additional functional activities do you think should be included in the PEDI-CAT Wheelchair subdomain item bank? (Therapists were shown the existing Wheelchair Sub-domain items).

3. There are no power wheelchair items in the Wheelchair Sub-domain. Would power wheelchair items be helpful to you? What are some examples of skills/items that you would envision?

Answers to focus group questions and ideas for items were noted during the groups on white boards and photographed. In addition, the UK group discussions were audio-recorded. These recordings were immediately destroyed following transcription.

Following the focus groups, a draft set of items was produced by this study’s investigators. These items were then evaluated through cognitive interviews with physical therapists (Willis, 1999). The procedures for the cognitive interviews included asking the therapists to read a test item and to verbally explain to the interviewer what they understood the item to mean (e.g., “In your own words, please describe what this item is asking.”). Directed probes to further explore definitions or concepts within a test item (e.g. meaning, wording) and “think aloud” strategies in which respondents read an item and verbally explain their thought process while contemplating the meaning of the test item were used. Response choices for each item were also probed (Dumas et al., 2008). This occurred in a cyclical round with review and refinement of items by the study authors after

each interview (n= 17 total). Following this, the last draft item set was then reviewed by the study authors for duplication, clarity, and breadth of content, and a preliminary new PEDI-CAT item pool was produced.

Data Analysis

All data analysis was completed by the study investigators, three physiotherapists with more than 30+ years of experience working with children with disabilities and using, evaluating and/or developing functional outcome measures. Data from focus groups was recorded, transcribed, and analysed to interpret meaning from the content of the focus group data. Study authors, ultimately categorised potential new items by type of wheelchair users (e.g. manual-self propel, manual-dependent, power wheelchair user). All potentially new items were kept at this stage even if not appearing in all data collection events, to ensure data saturation.

After each cognitive testing session was complete, investigators compiled handwritten feedback regarding each individual item and its response options. Depending on the outcome of interview questions and the data obtained, subsequent interview questions were amended if it was thought this would produce more specific data. This proceeded in a cyclical format. This feedback was then separated and again coded by type of wheelchair (manual, power) and PEDI-CAT response option. Constant comparative analysis was used for cross-validation among interviews as concerns and questions identified by each participant were compared with those expressed by other therapists. After each set of interviews, investigators refined items for clarity and tested the revised items in subsequent interviews. Once no new information for any item was being generated in the interviews, this stage was terminated and a preliminary set of new items produced. Items considered irrelevant (e.g. content covered in other items or the content was only relevant to a few children) were removed. Items were added if important areas were missing.

Results

Our literature review yielded 9 wheelchairs skills tests with content relevant to children and young people with disabilities. The nine measures were as follows: the Wheelchair Skills Test for manual and power wheelchair users (WST) (Lindquist et al., 2010; Rushton et al., 2016), Wheel-Con (Sakakibara et al., 2015), Powered Mobility Screening Tool (Kamaraj et al., 2014), Powered Mobility Checklist (Dunaway et al., 2013), Wheelchair Outcome Measure (WHOM) (Mortenson et al., 2007), Functioning Everyday in a Wheelchair (FEW) (Mills et al., 2007), Tufts Assessment of Motor Performance (Haley & Ludlow, 1992), Stages in Operating a Joystick (Nilsson et al.,

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2011) and Durkin’s Tool of Assessment (Nilsson & Durkin, 2014). Only the WST, Wheel-Con and FEW had evidence of strong reliability and validity. Tests were both judgement based and capacity measures.

Constructs from the identified measures included: propulsion (self and by others), distance, surfaces, transfers, turning, sprinting, wheelies, indoor and outdoor skills, community participation, wheelchair management (folding and unfolding), basic cause and effect, directional control, negotiating obstacles and confidence in wheelchair skills. None of the tests identified covered all the skills associated with wheelchair use in children with disabilities. They also lacked sufficient easier items to have wider application to younger children and children with more severe mobility impairments, such as maintaining head control while being pushed in a wheelchair, moving in a wheelchair for adjustment of clothing or insertion of slings, and placing feet onto foot rests.

Therapists participating in the focus groups and cognitive interviews had a range of experience working with children (range 1-30 years). All therapists had children with disabilities on their caseloads, including younger children < 3 years, and children with more severe motor impairments (GMFCS Level IV-V), as this was necessary for having the knowledge of which items might be appropriate for such client groups.

The three focus groups generated 83 potential new items with a subset of items which were common to all wheelchair users (n=51), items specific to children who self-propelled (n=14) and other items specific to children driving power wheelchairs (n=18). Examples are given in Table 1.

The first round of cognitive interviews included 4 USA and 3 UK PTs, each of which reviewed 20-68 items, in one or two interviews (total interviews = 8). After the first round of cognitive interviews (n=8), 85 wheelchair items were produced: all wheelchair users (n=48), only manual self-propelling chair users (n=18) and only powered wheelchair users (n=19). Following review of this data, items were refined and a second round of interviews took place (USA, n=3; UK, n=6), each PT reviewing 42 to 53 items each. After the second round of cognitive interviews (n=9), wheelchair items increased to 115 but once all interviews were completed, 99 items remained: all wheelchair users (n=33), only manual (n=36) and only powered (n=30). No differences in item content were observed when comparing data from therapists in the USA and UK. Only minimal differences in wording (e.g. elevator versus lift) or in spelling were noted.

An example follows showing the process of how one wheelchair item was amended from an idea generated from a focus group, through the cognitive

interviewing process:

Initial draft of item from focus group data: ‘Accesses pen, smart phone, something from back of chair’.

1st Interview: Suggested making specific to a backpack: ‘Manages a backpack on manual wheelchair (places on/off wheelchair handles, can put in/take out objects)’

2nd Interview: Divided into 2 items to improve clarity of potential responses:

• ‘Reaches bag or backpack on wheelchair’

• ‘Places bag or backpack on/off wheelchair handles’

Final Interview: Suggested not just reaching the bag/backpack but making it more functional – reaching into bag (for an item).

• ‘Reaches into bag or backpack on wheelchair’

• ‘Places bag or backpack on/off wheelchair handles’

Examples of items following the final round of cognitive interviews are shown in Table 2.

During cognitive interviewing, the relevance of some items and/or the response options were often interpreted differently by individual therapists. Examples include the item, “Press joystick, switch or other powered wheelchair controls momentarily”. One therapist thought this was clear, it would demonstrate the ‘child knows what happens when they push the button’ and it would thus be easy to select a response on the scale. A second therapist felt this skill did not suggest the child was at the stage of ‘using’ a power chair, considered that it would be difficult to score and should be omitted. A third therapist interpreted ‘Easy’ as the child could keep their hand down; 'hard' as needing increased time to get hand on, a hand to assist, or part of body was compensating so it would be deemed effortful, but was less sure of scoring if the child was activating the switch by pushing into extension. Overall, the ability to operate the controls was considered sufficiently relevant to retain, so it was amended to be deemed more purposeful: “Presses and holds power wheelchair controls (e.g. joystick) for a few seconds to move in any direction”. Response options for the PEDI-CAT are however already defined, and are generic with descriptors, and were not changed.

Examples of other items where there was inconsistency in interpretation of responses include: propelling or driving a chair up and down a ramp; propelling or driving a chair over different distances; propelling or driving a wheelchair outdoors on cracked or uneven paved surfaces; stopping a manual wheelchair on command; and some of the items relating to maintaining head control while in a wheelchair for


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specific times.

Discussion

The purpose of this study was to develop a new preliminary item pool for the PEDI-CAT Wheelchair subdomain for children who self-propel manual wheelchairs, drive powered chairs or are pushed in a wheelchair. This was in response to recent research indicating limitations with the PEDI-CAT’s current Wheelchair subdomain. A literature review indicated limited options of measures which could be used by physiotherapists working with children with a wide range of disabilities, and who may want to measure improvement with wheelchair skills as a therapeutic outcome.

The methods for this study (literature review, focus groups and cognitive interviews) modelled the procedures used in previous work to develop items for the PEDI-CAT (Dumas, 2010). The process of using focus groups for generation of item contents proved effective, and is a tried and tested method for development of patient reported outcome measures (FDA, 2009; Mokkink et al., 2010; Patrick et al., 2011; Reeve et al., 2013). To reach data saturation within cognitive interviewing, when no new comments on the proposed items were appearing took >17 hours of interviews. This resulted in a potential item pool of 99 items to fully cover the test construct. This could be considered a burden for parents to complete. In construction of tests, it is considered important to ensure the content of questionnaires is comprehensive and relevant to users (Rolstad et al., 2011). A computer adaptive test such as the PEDI-CAT allows for reduction of items with item response scaling, yet ensures relevance.

During cognitive interviews, there was extensive discussion with therapists about whether the type of power chair controls should be specified. Some therapists thought this was very important, but the majority thought there were too many variations to include all of them as individual items. To measure endurance, some therapists favoured items specifying the distance and some preferred items including the specific time children could self-propel or drive their powered wheelchair. Eventually therapists concluded that these were not interchangeable and both needed to be included, but with more emphasis on items focusing on distance. For example, one child might self- propel 500 metres quite quickly, but be unable to keep going for a full 15 minutes, whereas another child might not manage 500 metres but be able to self-propel slowly for 15 minutes.

Existing items on the PEDI-CAT include pictures to aid clarity of items and it is anticipated that the new items will be accompanied by pictures (H. Dumas et al., 2010) . During cognitive interviews which

included therapists unfamiliar with the PEDI-CAT, therapists frequently concluded that the wording of some items could not clearly convey the intended activity without a picture.

Therapists found it difficult to be precise in selecting response options due to the generic PEDI-CAT response scale which includes several descriptors at each level (H. Dumas et al., 2010) . For example, ‘a little hard’ is defined as ‘Does with a little help, extra time or effort’. In the cognitive interviews, if therapists were asked to describe what they thought ‘a little hard’ represented for an item, they interpreted the response options in a variety of ways for the same items. If therapists were asked to think of a child on their caseload and try to give them a score, some found it difficult. For example, the child might take a lot of extra time but need no help. Determining the appropriate response is more difficult when several descriptors exist for one response level. This can also ultimately reduce the information available to a clinician as they do not know which factor has changed when a score changes. Therefore the PEDI-CAT response scales are to be reviewed in a future stage of this research to reduce response error.

This new preliminary set of items will be tested with parents to further establish content validity. It is anticipated that some items may be removed at this stage as it is likely that not all items will be considered relevant by parents. The final items will then be subject to item response theory (IRT) analysis. This produces an interval level scale of items from easy to difficult. This allows the clinician to identify exactly where a child is functioning on a scale and the next most difficult items thus helping with goal setting, treatment planning and in providing feedback to families (Russell et al., 2014). In addition, IRT can specify how well different items describe the group of children and how well individual children fit the group. Items which do not measure something distinctly different to other items are removed, as retaining them can lead to redundancy and decreased precision(Hays et al., 2000).

The PEDI-CAT is a computer adaptive test (H. Dumas et al., 2010), requiring only the minimum number of items necessary to produce a valid test result. Therefore, despite having a proposed set of up to 99 items at this stage in the research, a far smaller number of items will be used per assessment. Currently, the PEDI-CAT’s 4 domains contain 276 items but only 15-30 items per domain need to be administered to obtain an accurate and precise score, taking an average of 12 minutes in total (Dumas, Fragala-Pinkham, et al., 2012).

The limitations to this study are that only therapists in the UK and USA were included in the development and refinement of test items, so these items may not


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cover all skills or be all relevant to therapists in other countries. As purposive convenience samples were used, it is possible that other therapists and/or a sample representing a wider population of therapists might have produced different ideas for potential items. However, within this sample, data saturation for potential test items was reached. The items have not yet been tested on the ultimate users of the measure, parents of children with disabilities, so are in a preliminary stage.

Conclusion

Through a literature review, a series of focus groups and cognitive interviews with USA physical

therapists and UK physiotherapists, a preliminary set of 99 new wheelchair items have been developed for the PEDI-CAT. This will potentially broaden the PEDI-CAT to include important skills for all children with disabilities who either self-propel a manual wheelchair or drive a power wheelchair.

In subsequent phases of research, the response options and this preliminary item pool will be reviewed by parents and therapists, the item pool will be refined, and the item bank will be calibrated on a large sample of children who use wheelchairs. Finally, the data from the calibration sample will be subject to an IRT analysis to produce a final set of items and the computer adaptive test algorithm will be developed.


Table 1: Examples of potential items from focus groups

Examples of potential skills relevant to both manual & power wheelchair users

Relieve pressure by shifting side to side

Carry objects, e.g. on tray

Manage a backpack

Direct others to undo seatbelt, move footplates etc.

Keep arm(s) in e.g. when go through doorway

Reach for something while in chair, e.g. switch, get something in classroom

Take coat off / put coat on

Examples of potential manual wheelchair skills items

Move forwards

Move backwards

Keep within lines going forwards, or within a track

Control speed down a hill

Propel to/from school

Negotiate in classroom

Negotiate around playground

Propel for a school day

Examples of potential power wheelchair skills items

Turn controls on/off

Stop on command when appropriate

Adjust speed appropriately: slow down as approach bed/chair for transfers

Pick up tray at lunchtime and take to table

Control tilt, high/low /stand up features

Get in/out of a bus

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Table 2: Examples of PEDI-CAT items after cognitive interview process

Manual wheelchair

Puts brakes on while in manual wheelchair

Propels manual wheelchair him/herself on rugs and carpet

Propels manual wheelchair him/herself 5 metres in wide hallway or open indoor space

Propels manual wheelchair him/herself around home or classroom furniture

Propels manual wheelchair him/herself quickly indoors to keep up with another person or respond to a fire drill

Propels manual wheelchair him/herself up to desk, table or sink

Manoeuvres manual wheelchair him/herself up ramp to enter home or building

Propels manual wheelchair 50-100 feet/15-30 meters on paved surfaces (approximately 1 city block)

Powered Wheelchair

Presses and holds power wheelchair controls (e.g. joystick) for a few seconds to move in any direction

Lifts and turns head to look in direction of travel while driving power wheelchair

Drives power wheelchair short distance (15 feet/5 meters) down hallway in straight line

Drives power wheelchair at appropriate speed for setting such as crowded classroom

Operates power wheelchair seating functions (e.g. recline and tilt)

Drives power wheelchair up to desk, table or sink

Scoots forward in wheelchair in preparation for transfer

Transfers from wheelchair to adult-sized toilet

Transported in Wheelchair and/or physically dependent for transfers

Typically lifts head momentarily while sitting in wheelchair (e.g. in response to name being called or seeing a familiar person)

Typically keeps head against headrest for 10 minutes (without flopping down to sides), while being pushed in reclined or tilted wheelchair

Typically keeps head up for 10 minutes while being pushed in upright wheelchair, may use headrest for support

Brings head forward in wheelchair to assist with dressing or positioning

Leans upper body forward in wheelchair to assist with dressing or positioning

Moves side to side in wheelchair for pressure relief or to position sling for lift

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References

Artino, A. R., La Rochelle, J. S., Dezee, K. J., & Gehlbach, H. (2014). Developing questionnaires for educational research: AMEE Guide No. 87. Medical Teacher, 36(6), 463-474.

Dumas, H., Fragala-Pinkham, M., & Haley, S. (2010). Development of a postacute hosptial item bank for the new Pediatric Evaluation of Disability Inventory-Computer Adaptive Test. Int J of Rehabil Research, 33(4), 332-338.

Dumas, H., Fragala-Pinkham, M., Haley, S., Coster, W., Kramer, J., Kao, Y. C., & Moed, R. (2010). Item bank development for a revised pediatric evaluation of disability inventory (PEDI). Phys Occup Ther Pediatr, 30(3), 168-184.

Dumas, H. M., & Fragala-Pinkham, M. A. (2012). Concurrent validity and reliability of the pediatric evaluation of disability inventory-computer adaptive test mobility domain. Pediatr Phys Ther, 24(2), 171-176; discussion 176.

Dumas, H. M., Fragala-Pinkham, M. A., Haley, S. M., Ni, P., Coster, W., Kramer, J. M., Kao, Y. C., Moed, R., & Ludlow, L. H. (2012). Computer adaptive test performance in children with and without disabilities: prospective field study of the PEDI-CAT. Disability and Rehabilitation, 34(5), 393-401.

Dumas, H. M., Watson, K., Fragala-Pinkham, M. A., Haley, S. M., Bilodeau, N., Montpetit, K., Gorton, G. E., 3rd, Mulcahey, M. J., & Tucker, C. A. (2008). Using cognitive interviewing for test items to assess physical function in children with cerebral palsy. Pediatr Phys Ther, 20(4), 356-362.

Dunaway, S., Montes, J., O'Hagen, J., Sproule, D. M., Vivo, D. C., & Kaufmann, P. (2013). Independent mobility after early introduction of a power wheelchair in spinal muscular atrophy. J Child Neurol, 28(5), 576-582.

FDA. (2009). Guidance for industry patient-reported outcome measures: use in medical product development to support labeling claims Retrieved from https://www.fda.gov/downloads/drugs/guidances/ucm193282.pdf website: www.fda.gov

Fragala-Pinkham, M., Dumas, H., Lombard, K., & O'Brien, J. (2016). Responsiveness of the Pediatric Evaluation of Disability Inventory - Computer Adaptive Test in measuring functional outcomes for inpatient pediatric rehabilitation. J of Pediatr Rehabil Med, 9(3), 215-222.

Haley, S., Coster, W., Dumas, H., Fragala-Pinkham, M., & Moed, R. (2012). PEDI-CAT: Development, Standardization and Administration Manual Boston, MA: CRECare, LLC.

Haley, S. M., Coster, W. J., Dumas, H. M., Fragala-Pinkham, M. A., Kramer, J., Ni, P., Tian, F., Kao, Y. C., Moed, R., & Ludlow, L. H. (2011). Accuracy and precision of the Pediatric Evaluation of Disability Inventory computer-adaptive tests (PEDI-CAT). Developmental Medicine & Child Neurology, 53(12), 1100-1106.

Haley, S. M., & Ludlow, L. H. (1992). Applicability of the hierarchical scales of the Tufts Assessment of Motor Performance for school-aged children and adults with disabilities. Phys Ther, 72(3), 191-202; discussion 202-196.

Hays, R. D., Morales, L. S., & Reise, S. P. (2000). Item Response Theory and health outcomes measurement in the 21st Century. Medical care, 38(9 Suppl), II28-II42.

Kamaraj, D. C., Dicianno, B. E., & Cooper, R. A. (2014). A participatory approach to develop the Power Mobility Screening Tool and the Power Mobility Clinical Driving Assessment tool. Biomed Res Int, 2014, 541614.

Lindquist, N. J., Loudon, P. E., Magis, T. F., Rispin, J. E., Kirby, R. L., & Manns, P. J. (2010). Reliability of the performance and safety scores of the wheelchair skills test version 4.1 for manual wheelchair users. Arch Phys Med Rehabil, 91(11), 1752-1757.

Mills, T. L., Holm, M. B., & Schmeler, M. (2007). Test-retest reliability and cross validation of the functioning everyday with a wheelchair instrument. Assist Technol, 19(2), 61-77.

Mokkink, B., Terwee, C., Patrick, D., Alonso, J., Stratford, P., Knol, D., Bouter, L., & de Vet, H. (2010). The COSMIN checklist for assessing the methodological quality of studies on measurement properties of health status measurement instruments: an international Delphi study. Qual Life Res, 19(4), 539-549.

Mortenson, W. B., Miller, W. C., & Miller-Pogar, J. (2007). Measuring wheelchair intervention outcomes: development of the wheelchair outcome measure. Disabil Rehabil Assist Technol, 2(5), 275-285.

Nilsson, L., & Durkin, J. (2014). Assessment of learning powered mobility use - applying grounded theory to occupational performance. J Rehabil Res Dev, 51(6), 963-974.

Nilsson, L., Eklund, M., & Nyberg, P. (2011). Driving to learn in a powered wheelchair: inter-rater reliability of a tool for assessment of joystick-use. Aust Occup Ther J, 58(6), 447-454.

Patrick, D. L., Burke, L. B., Gwaltney, C. J., Leidy, N. K., Martin, M. L., Molsen, E., & Ring, L. (2011). Establishing and reporting the evidence in newly developed patient-reported outcomes (PRO)

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Instruments for medical product evaluation: ISPOR PRO Good research practices task force report: Part 1: Eliciting concepts for a new PRO instrument. Value Health, 14(8), 967-977.

Reeve, B. B., Wyrwich, K. W., Wu, A. W., Velikova, G., Terwee, C. B., Snyder, C. F., Schwartz, C., Revicki, D. A., Moinpour, C. M., McLeod, L. D., Lyons, J. C., Lenderking, W. R., Hinds, P. S., Hays, R. D., Greenhalgh, J., Gershon, R., Feeny, D., Fayers, P. M., Cella, D., & Brundage, M. (2013). ISOQOL recommends minimum standards for patient-reported outcome measures used in patient-centered outcomes and comparative effectiveness research. Qual Life Res, 22(8), 1889-1905.

Rolstad, S., Adler, J., & Rydén, A. (2011). Response burden and questionnaire length: Is shorter better? A review and meta-analysis. Value Health, 14(8), 1101-1108.

Rushton, P. W., Kirby, R. L., Routhier, F., & Smith, C. (2016). Measurement properties of the Wheelchair Skills Test-Questionnaire for powered wheelchair users. Disabil Rehabil Assist Technol, 11(5), 400-406.

Russell, D., Rosenbaum, P., Wright, M., & Avery, L. (2014). Gross Motor Function Measure (GMFM-66 & GMFM-88) User's Manual (Second ed.). London: MacKeith press.

Sakakibara, B. M., Miller, W. C., & Rushton, P. W. (2015). Rasch analyses of the wheelchair use confidence scale. Arch Phys Med Rehabil, 96(6), 1036-1044.

Walsh, T. R., Irwin, D. E., Meier, A., Varni, J. W., & DeWalt, D. A. (2008). The use of focus groups in the development of the PROMIS Pediatrics Item Bank. Qual Life Res, 17(5), 725-735.

Willis, G. (1999). Cognitive Interviewing - a how to guide. Retrieved 08.01.17., 2017, from www.hkr.se/contentassets/9ed7b1b3997e4bf4baa8d4eceed5cd87/gordonwillis.pdf

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Descriptions of Rare Diseases Relevant to Paediatric PhysiotherapyA disease or disorder is defined as rare in Europe if it affects less than 1 in 2,000. One rare disease may

affect only a handful of patients in the EU, and another touch as many as 245,000. There are between 6,000 and 8,000 rare diseases. On the whole, rare diseases may affect 30 million European Union citizens. Eighty

percent of rare diseases are of genetic origin, and are often chronic and life-threatening. European Alliance for Rare Diseases

www.eurordis.org It is estimated that up to 4 million children and adults are affected by rare disorders in the UK according to Contact a Family

www.cafamily.org.uk

Jeune Syndrome

Jeune syndrome was identified by Jeune et.al. in 1955. It is an autosomal recessive disease also known as asphyxiating thoracic dystrophy (Buget et.al 2015).

Jeune syndrome is a skeletal dysplasia categorised by a narrow thoracic cage and a variety of skeletal and extra skeletal abnormalities. The syndrome is known to be genetically heterogeneous; at least 9 genes have been identified (Shaheen et.al. 2015).

IncidenceJeune syndrome is a very rare disease and is estimated to occur in 1 per 100,000-130,000 live births (Ramya et.al. 2013).

Males, females, race and ethnic groups are equally affected.

Diagnosis can be determined before birth if characteristic signs and symptoms are present on ultrasound. Clinical findings and radiography confirm diagnosis postnatally, but it can be confirmed by testing for mutations in genes (Miller 2015).

Differential diagnosis includes Ellis van Creveld syndrome which has similar features, although the respiratory system is not as severely affected and kidney failure is not a characteristic (de Vries et al 2010).

PrognosisSurvival has been reported to the 4th decade of life (O’Connor et.al. 2008).

The severity of the thoracic deformity is the main prognosis factor and fatality rates in early childhood are as high as 60-80%. Respiratory distress tends to decrease with age potentially due to the improved mechanical properties of the chest wall with growth. Renal issues can develop as the child gets older (de Vries et.al. 2010).

Respiratory distress in children under 2 and renal failure in children aged 3-10 are the two primary causes of death (Keppler-Norueil et.al. 2011).

Clinical featuresThe core phenotype involves a small, narrow thoracic cage, with short ribs which often results in respiratory

problems and asphyxiation.

Skeletal features include short horizontal ribs, small statue, pectus carinatum, polydactyly, spinal curvature abnormalities including scoliosis, kyphosis or both and short limbs (Miller 2013).

Non skeletal features include liver and kidney disease and frequent respiratory infections (O’Connor et.al. 2008).

Physiotherapy involvementLiterature and evidence related to Jeune syndrome is currently very limited.

During the first years, dependant on severity, respiratory complications due to chest deformities and thoracic involvement are the main problem in Jeune syndrome (de Vries et.al. 2010).

Respiratory distress and recurrent infections can be common during the neonatal period and infancy. Intensive care maybe considered dependant on severity (Saito et.al. 2016).

Physiotherapy input either in a hospital setting or a community environment includes treatment for recurrent respiratory infections with or without mechanical ventilation (Davies et.al. 2001).

Physiotherapy treatment is symptomatic dependant on clinical presentation and severity (deVries et.al. 2010). Manual techniques include vibrations, percussion and suction during respiratory infections to remove secretions and improve ventilation (Taussig & Landau 2008).

In some, the restrictive lung disease is sufficiently severe that treatment may include intubation, ventilator support and tracheostomy intervention.

Long term tracheotomy may allow for both survival and improvement of overall pulmonary function (Pauli 2009).

As the respiratory intervention is focused on patients at such a young age, advice & support is provided to parents and caregivers (Oberwaldner 2000).

Advice on positioning is also important during treatment, sleeping and feeding to optimise ventilation (Bertone 1988).

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Collaborative working with Occupational Therapy to provide equipment to reduce any spinal abnormalities and promote optimal positioning maybe required dependant on severity.

It is recommended that patients are frequently seen by a specialised paediatrician in a tertiary centre. After 2 years old, depending on their clinical condition, this can be reduced. Spirometry is advised yearly from the age of 5 (de Vries et.al. 2010). Communication between the members of the MDT is essential to promote a holistic approach to manage children and young people with Jeune Syndrome.

It has also been identified that some patients need monitoring for sleep-related hypoventilation after infancy (Leroy et.al. 2010). These include symptoms of severe fatigue, headaches and fear of going to sleep (Miller 2013). The use of non-invasive ventilation has been successfully used with sleep-related hypoventilation (Marcus 2001).

Surgical interventionLateral thoracic expansion (LTE) is a surgical procedure to create an enlarged chest wall. Evidence has demonstrated this is a safe and effective procedure for patients with Jeune syndrome older than 1 year old (Davies et.al. 2001).

Recently vertical expandable prosthetic titanium rib (VEPTR) has been applied to manage scoliosis and/ or lethal respiratory condition in infantile Jeune syndrome patients (Saito et. al. 2016).

SummaryJeune syndrome is a very rare condition with a variety of clinical features. Respiratory severity is variable with a high percentage of fatalities under 2 years old.

Physiotherapy treatment is dependent on severity and individual presentation to provide support in both a hospital environment and in the community setting.

Joanne Thornton Specialist Paediatric Physiotherapist Rainbows Centre, Burnley Hospital

References

Bertone N. (1998) Physiotherapy in a Neonatal Intensive Care Unit. The Australian Journal of Physiotherapy Vol.34 No. 1

Buget MI, Ozkan E, Edipoglu IS & Kucukay S (2015) Anesthetic Approach for a patient with Jeune Syndrome. Case reports in Anesthesiology. Vol 2018 Article 509196

Davies JT, Heistein JB, Castile RG, Mutabagi KH, Villalobos RE & Ruberg RL (2001) Lateral thoracic expansion for Jeune’s syndrome: Mid term results. Ann Thorac Surg 72: 872-8

de Vries J, Yntema JL, van Die CE, Crama n, Comelissen EAM & Hamel CJ (2010) Jeune syndrome: description of 13 cases and a proposal for a follow up protocol. Eur J Paediatr. 169: 77-88

Jeune M, Bèraud C & Carron R (1955) Dystrophie thoracique asphyxiante de caractère familial. Archives of Franç de Pediatrie. Vol 12 pp. 886-891

Keppler-Noreuil KM, Adam MP, Welch J, Muilenburg A & Willing MC. (2011) Clinical insights gained from eight new cases and review of reported cases with Jeune syndrome (asphyxiating thoracic dystrophy). Am J Med Genet A. May; 155A(5):1021-32

Leroy P, Martens M & Schott N (2010) Late respiratory failure in Jeune syndrome. Eur J Paediatr 169: 375-376

Marcus CL. (2001) Sleep-disordered Breathing in Children. American Journal of Respiratory and Critical Care Medicine Vol. 164, No. 1

Miller KA (2013) Jeune Asphyxiating Thoracic Dystrophy, Forgotten Disease website. http://www.forgottendiseases.org/assets/jeunesyn.html Viewed 24/10/17

Oberwaldner B. (2000) Physiotherapy for airways clearance in Paediatrics. Eur Respir J 15: 196-204

O’Connor MB, Gallagher DP & Mulloy E (2008) Jeune syndrome. Post grad Med J Vol. 84 No. 996

Pauli RM. (2009) Jeune Asphyxiating thoracic dystrophy (Jeunes syndrome) Natural history. Midwest Regional Bone Dysplasia Clinics. http://www.lpaonline.org Viewed 31/01/18

Ramirez N, Villarin S, Ritchie R & Thompson KJ (2015) Thoracic Insufficiency Syndrome: An Overview. Razavi Int J Med. 2015; 3 (4):e330330

Ramya HS, Sushanth S & Manjunath MN (2013) Jeune syndrome. BMJ Vol 6 No 2

Saito W, Inoue G, Imura T, Nakazawa T, Miyagi M, Namba T, Shirasawa E, Takahira N & Takaso M (2016) Spinal Correction of Scoliosis in Jeune Syndrome: a report of two cases. Scoliosis & Spinal Diseases 11:7

Shaheen R, Schmidts M, Faqeih E, Hashem A, Lausch E, Holder I, Superti-Fuaga A, Mitchison HM, Almoisheer A, Alamo R, Alshiddi T, Alzahrani F, Beales PL & Alkuraya FS (2015) A founder CEP120 Mutation in Jeunes asphyxiating thoracic dystrophy expands the role of centriolar proteins in skeletal ciliopathies. Human Molecular Genetics Vol. 24 No 5 1410-1419

Taussig L & Landau L. (2008) Pediatric Respiratory Medicine. 2nd Edition. Philadelphia, Mosby Pg. 241-243

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Summary of NICE Guideline for Developmental Follow-up of Children and Young People Born Preterm

NICE guideline NG72 August 2017

Jade Kant- Neurodevelopmental Physiotherapist, Leeds Teaching Hospital NHS TrustHilary Cruickshank- Neonatal Specialist, Edinburgh Hospital NHS Trust

Author’s email: [email protected]

Background

These guidelines were published in August 2017 and are widely accepted as the minimal standard the services should offer for follow-up. The guideline is specifically for follow-up of babies, children and young people under the age of eighteen years, who were born before 37 weeks gestation.

The guideline was produced to help improve the identification of developmental problems and concern by setting standards for follow-up. The implications for physiotherapists working in this field and for future planning services will be summarized.

Summary of Recommendations

Information and support for parents and carers of preterm babies

Provide information for parents and carers on:

• Risk and prevalence of developmental problems and disorders

• Information tailored to their individual circumstances taking into account potential development, level of education, social needs, and cultural, spiritual or religious belief. Be consistent in information sharing among healthcare professionals.

Follow the principles in NICE guidelines (www.nice.org.uk/guidance/cg138)in relation to communication, discharge planning. Start discharge planning as soon after preterm birth as possible.

Help parents or carers to gain knowledge, skills and confidence.

Provide emotional and psychological support.

Provide information about opportunities for peer support.

Inform parents or carers about routine postnatal care and support available.

Explain about universal services and national recommendations for assessing the development of the child. Discuss whether their baby will be offered enhanced developmental support.

Explain about corrected age and the child’s

development. If they have any concerns regarding their child’s development they can go to health visitor or GP.

Professionals providing the postnatal care and support should have the skills and knowledge to recognize and manage any problems.

Risk and prevalence of developmental problems and disorders

Be aware that children and young people born preterm have an increased risk of developmental problems and disorders. There was an absence of evidence about overall risk and prevalence in children born preterm, along with limited evidence about developmental concerns in 11 to 18 year olds. At times the evidence was underpowered to detect an effect and gestational ages were unable to be extrapolated at times.

Cerebral palsy – preterm children have an increased risk of developing CP, but other independent risks are grade 3 or 4 intraventricular haemorrhage (IVH), cystic periventricular leukomalacia (PVL), neonatal sepsis, bronchopulmonary dysplasia which needed mechanical ventilation at 36 weeks post menstrual age, antenatal steroids, postnatal steroids for babies born after 32 weeks gestation, prevalence increases with decreasing gestational age.

Motor function problems - preterm children have an increased risk of developing motor function problems, but other independent risks are grade 3 or 4 IVH, PVL or infarct, necrotizing enterocolitis (NEC) that required surgery, neonatal sepsis and severe retinopathy of prematurity.

There is increased risk compared to the general population of developmental coordination disorder.

Learning disability - preterm children have an increased risk of developing motor function problems, but other independent risks are grade 3 or 4 IVH, PVL or neonatal sepsis before 28 weeks gestation, NEC needing surgery before 33 weeks gestation, bronchopulmonary dysplasia still needing mechanical ventilation at 36 weeks post menstrual age in babies born before 28 weeks gestation, severe ROP for babies born before 28 weeks gestation, small for gestational age, postnatal steroids given to babies

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born before 33 weeks gestation, mother from a low-income disadvantage background and prevalence increases with decreasing gestational age.

Special educational needs and educational attainment – children born preterm have an increased risk of needing specialist education and brain lesions detected by ultrasound or being male are considered extra increased risk factors. These children are at risk of low educational attainment in key stage 1. Preterm babies born before 26 weeks gestation have a greater risk of low attainment in reading and mathematics. Independent risk factors are IVH or bronchopulmonary dysplasia for which mechanical ventilation at 36 weeks postmenstrual age was needed.

Executive function problems are an increased risk if born before 32 weeks gestation.

Speech, language and communication disorders and problems are a risk if born preterm and independent risk factors are, grade 3 or 4 IVH, cystic PVL or being male.

Attention, impulsivity and hyperactivity are an increased risk if born before 33 weeks gestation. If born before 28 weeks gestation there is an increased risk of attention deficit hyperactivity disorder (ADHD) and being male is an independent risk factor.

Autism spectrum disorder – being born preterm is an increased risk factor, but independent risk factors are being male or IVH before 34 weeks gestation. If born before 28 weeks gestation there is an increased risk of symptoms of social communication impairment, which may suggest a problem on the autistic spectrum.

Emotional behavioural problems – if born preterm there is this increased risk, but other independent risk factors are major brain lesions, mother with mental health problems, mother younger than 25 years, mother from a low income or disadvantaged background.

Feeding problems – preterm babies have an increased risk of oro-motor feeding problems and this risk can persists until 6 years old in children born before 26 weeks gestation.

Sleep problems – there is an increased risk of sleep apneas up to the age of 6 years old if born preterm.

Visual impairment – the prevalence of visual impairment increases with decreasing gestational age. Independent risk factors are grade 3 or 4 IV with a shunt, neonatal sepsis in babies born before 33 weeks gestation or ROP needing treatment.

Hearing impairment - the prevalence of visual impairment increases with decreasing gestational age and neonatal sepsis is an independent risk factor if

born before 28 weeks gestation.

Developmental delay – is a risk if born preterm. Independent risk factors are small for gestational, being male, mother from a low-income or disadvantaged background, black, Asian or other minority ethnic group or multiple pregnancy.

Other gestational ages and factors might be associated with increased risk of developmental problems, but might have been listed.

Enhanced developmental support and surveillance

Criteria for enhanced developmental support and surveillance up to 2 years corrected age:

Provided by a multidisciplinary team for children who have a developmental problem or disorder, or an increased risk of developing one, if born before 30 weeks gestation, between 30 and 36+6 weeks gestation and has one or more of the following - a brain lesion on neuroimaging associated with developmental problems or disorders, grade 2 or 3 hypoxic ischaemic encephalopathy in neonatal period, neonatal bacterial meningitis or herpes simplex encephalitis in neonatal period. Preterm infants suspected of increased developmental problems or disorders.

Criteria for enhanced developmental support at age 4 years uncorrected – face to face developmental assessment at this age for all children born before 28 weeks gestation.

Enhanced support should provide parents or carers with a single point of contact after discharge, use a range of approaches and be individual, such as face to face meetings, in clinics or at home, a telephone helpline or text messages are a few examples.

Enhanced developmental surveillance for up to 2 years olds corrected, should have the minimum of 2 face to face follow-up visits in the first year, which focus on development at corrected ages of between 3 and 5 months and by 12 months, along with detailed face to face developmental assessment at age 2 years corrected.

The professionals involved should have the appropriate skills and discuss if parents or carers have any developmental concerns, include checks for developmental problems and disorders, measure length or height, weight and head circumference, evaluate and review any developmental concerns, consider further investigations and refer to appropriate local pathway if needed.

At each enhanced developmental surveillance, signs of symptoms and problems of developmental concerns should be looked for, such as CP, global developmental delay and learning disability, autism spectrum disorder, visual impairment, hearing

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impairment, feeding problems, sleep problems including sleep apnea, speech, language and communication problems, motor problems, problems with inattention, impulsivity or hyperactivity, emotional and behavioral problems, executive function problems and potential special educational needs.

Recognise possible early motor signs of cerebral palsy, such as delayed motor milestones, unusual fidgety movements or other abnormalities of movement, including asymmetry or paucity of movement, abnormal tone, or persistent feeding difficulties.

Developmental assessment at 2 years uncorrected enhanced surveillance, should include, all aspects already listed above and using the PARCA – R (Parent Report of children’s Abilities – Revised, but if this is not suitable (eg because of poor English or outside the validated age range of 22 to 26 months), then use an alternative, GMFCS (Gross Motor Function Classification System score, if CP has been diagnosed, ensure vison and hearing checks along with national guidelines have been followed.

After the 2 year corrected age assessment, advise that their child should continue to be followed up by the universal screening services for all children and young people and if their child is born before 28 weeks gestation, report they will be offered a further developmental assessment at age 4 years uncorrected.

At age 4 years uncorrected, a face to face developmental assessment should be carried out if born before 28 weeks gestation, covering all previous aspects mentioned, but use the parent questionnaires of Strengths and Difficulties Questionnaire (SDQ) before the assessment and discuss during the assessment, as this checks for social, attentional, emotional and behavioural problems. Use the Ages and Stages Questionnaire (ASQ) 48-month questionnaire, to check for different aspects of development, review previous assessments, GMFCS score if CP diagnosed, check orthoptic screening and use a standardized test to assess IQ, such as the Wechsler Preschool and Primary Scales of Intelligence 4th Edition (WPPSI). Provide all this information in a comprehensive summary.

Share all the information gathered with parents or carers and local pathways. Ask for permission for sharing this information with education services and social services as appropriate. Later presentation of learning or behavioural problems can occur and this is why primary and secondary professionals should be aware of preterm births.

Delivering enhanced developmental support and surveillance

This should be provided for as part of the integral

neonatal service, working with local health services, empowering parents or carers, be delivered by an MDT and record outcomes at specific points.

The MDT professionals should have knowledge and expertise in neonatal care, development of children, provide support in community, administer and interpret results, collate information and know local care pathways. The professionals included should be neonatologists or paediatricians, nurses. For the 2 year check a neonatologist or paediatrician and at least one of occupational therapist, physiotherapist or speech and language therapist. For the 4 year check, an educational psychologist or clinical psychologist and a paediatrician. The MDT should have access to community nurses or health visitors, an occupational therapist, physiotherapist, speech and language therapist, a paediatric neurologist and a dietitian.

Neonatal audit

Record enhanced developmental surveillance results as applicable on the National Neonatal Research Database, any specialist neonatal care, reasons for enhanced surveillance, at the 2 years assessment, record a diagnosis, GMFCS, PARCA-R score, any epilepsy or impairments if hearing, vision, speech and language and motor skills. At the 4 year assessment record a diagnosis, GMFCS, full scale IQ score, SDQ total, any formal clinical diagnoses of developmental disorder, any epilepsy, and hearing impairment and results of orthoptic review.

Record any routine educational measures at Key stage 2.

Implications for practice for the paediatric/neonatal physiotherapist

These are widely accepted as the minimum standard for follow-up and many units currently use different tests and have different care pathways.

Any physiotherapist working in this area needs an in depth knowledge and skills to be able to work with this complex client group.

If your Trust decides to follow these guidelines, physiotherapists are well placed to be able to help complete many of these tests and be part of the MDT.

Currently many units do not follow-up to age 4 years and this may involve a change within units.

The full NICE guideline gives further details on these core areas.

Reference

www.nice.org.uk/guidance/ng72


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APCP Journal Volume 9 Number 1 (2018)

Submissions to the APCP Journal

The APCP Journal aims to disseminate original research, facilitate continuing medical education and to provide an opportunity to debate controversial issues in paediatric physiotherapy. Listed below are the different types of submissions that will be considered for publication in the APCP Journal with guidance on how to write and format your work to maximise the chances of your submission being successful. The Editorial Board also welcome pre-submission questions and will be happy to advise further. Please contact the Journal Editor – [email protected] for guidance or pre-submission advice.

Original Research Reports

Original research in many formats, including quantitative and qualitative research, case series and case reports. These articles should be 4,500 words or less, excluding the references and abstract. All submissions in this category will be subject to anonymous peer review by two reviewers.

Research Papers

Research papers should generally follow the ‘IMRD’ pattern (Introduction, Methods, Results and Discussion). When writing your paper, it is usually best to start with the most important parts, the methods and results, before writing the discussion, introduction and conclusion.

The methods section should include sufficient information to enable other clinicians to reproduce your work. Any work involving human or animal subjects must have appropriate ethical approval from the relevant Research Ethics Committee. In addition, written permission from children, parents or guardians to publish photographs of individuals must be submitted. The methods section should also include details of statistical methods if they are used and state which software, if any, was used to obtain the results. Any apparatus used in the study should be reported in terms of manufacturer and location (city, county, country).

The results section should be clear and easily understood. Rather than presenting the reader with masses of data, it often helps to construct your results to tell a story, taking the reader step by step through your findings. Do not present data twice in both text and tables/figures, and do not include material that belongs in the discussion, i.e. present results only, not interpretation. Consider how statistical data is presented, ensure that descriptive and inferential statistics are used appropriately to provide meaning to the data collected.

Tables and figures should be numbered consecutively as they are referred to in the text, and placed after

the references on a new sheet. Abbreviations should be explained in a footnote and only horizontal lines should be used. Table and figure captions/legends should be included on a separate sheet.

The discussion will allow you to succinctly summarise the major findings of your work and explain its relevance in terms of the available literature and current practice. It is helpful if the first paragraph briefly summarises the major findings. The discussion will also allow you to address any potential weaknesses in the methodology and justify why the research was performed in a particular way. It is important to keep the discussion relevant to the results obtained.

Ideally the introduction should be short and engage the reader, explaining why the paper is relevant to clinical practice. Often a brief summary of the existing literature highlighting the need for this particular research is useful, as it leads directly to the research question being asked.

Finally the title and abstract can be written. The abstract should be structured (limited to 300 words) consisting of ‘Background and Purpose’ (why the research was done), ‘Method’ (what was done), ‘Results’ (what was found) and ‘Conclusion’ (what was concluded). The title itself should describe the contents of the paper succinctly and accurately.

Scholarly Papers

Discursive papers sharing ideas or experiences in specific areas of practice can be structured more freely but should still include an ‘Introduction’, ‘Discussion’, and ‘Conclusions’. Scholarly papers should be no longer than 4,500 words.

Case Studies and Case Series

The format for case studies and case series differs from that given above and should start with an ‘Introduction’, followed by ‘Case Report’ (history, investigations, treatments, outcome), ‘Discussion’ and ‘Conclusion’. Case reports may be notable because they either focus on a rare condition or on a new method of treatment. The use of false names in case reports is encouraged but if a child is recognisable in the report (due to the condition or the specific nature of the treatment given), then written consent for publication should be obtained. Case reports should be no longer than 2,500 words.

Audit Reports

Reports of clinical audit should include an ‘Introduction’, ‘Standard Setting’ (with appropriate reference to the available literature), and ‘Method,

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Results’, ‘Discussion’, and ‘Conclusion’ these should be no longer than 3,000 words.

Review Papers

Systematic reviews undertake specific methodology and focus on a specific question, perform a thorough literature search and critical appraisal of individual studies using strict criteria. Less formal review articles will summarise the current literature on a particular topic. The Cochrane Collaboration has published a handbook on conducting systematic reviews (http://www.cochrane-handbook.org/) and you should structure your review in terms of ‘Introduction’, ‘Objectives’, ‘Methods’, ‘Results’, ‘Discussion’, and ‘Conclusion’.

There are published criteria that should be applied to the analysis of randomised controlled trials:

• the Delphi criteria

(http://www.ncbi.nlm. nih.gov/pubmed/10086815)

• the PEDro scale

(http:// www.pedro.org.au/scale_item.html)

• the MOOSE guidelines should be applied to the analysis of observational studies

(http://www.consortstatement.org/mod_product/uplo ads/MOOSE%20Statement%202000.pdf).

Review papers should be no more than 4,500 words.

Technical Evaluation

Technical evaluations describe mechanical or technical devices used in clinical practice or education and should include an ‘Introduction’, ‘Method’ including the specifications of the equipment used and the means of the evaluation, the ‘Evaluation’, ‘Discussion’ and ‘Conclusion’. Technical evaluations should be no longer than 2,500 words.

Service Development Report

A service development report should describe changes in service delivery/management. The structure of the report can be less formal but should include an introduction’, description of the service change(s), outcome and discussion on the implications for future practice. Service development reports should be no longer than 2,500 words.

Abstracts of Theses and Dissertations

Abstracts of research projects, audits and presentations from undergraduate and postgraduate degrees should be no more than 300 words in length and structured as a standard abstract (‘Introduction’, ‘Method’, ‘Results’, ‘Conclusion’). The Editorial Board would,

however, strongly encourage those considering such a submission to formulate their work instead as one of the above peer reviewed articles. In such cases, a pre-submission enquiry to the editor may be helpful.

Other types of editorial material

The Journal will also consider the following submissions:

• Letters to the editor

Letters to the editor can be on any issue pertinent to paediatric physiotherapy or to APCP. Letters should be no more than 500 words long.

• Book reviews

Book reviews should be no more than 750 words long.

Referencing

All work submitted for peer review should be referenced in the Harvard style:

In text, cite only the author(s) surname(s) followed by the date of publication, e.g. (Robinson, 1994) or Robinson (1994). ‘a’, ‘b’, etc., is used to indicate more than one publication by the same author(s) in the same year, e.g. 1992a,b). For three or more authors of a cited paper, name the first followed by et al, e.g. (Smith et al, 1990).

In the reference list, include articles in journals and books alphabetically by author. For citations from journals, give the names and initials of all authors (year of publication), title of the article, full name of the journal, volume number, issue number and first and last page numbers, e.g. Brown A, Green B and Gold C (2001). ‘The value of exercise’, Physiotherapy, 87, 1, 77-79. Referring to books, give the names and initials of all authors/editors (year of publication), title, publisher, place of publication, and the chapternumber or the page number of the citation or both, e.g. Gardner, M (2001). The Annotated Alice, Penguin Books, Harmondsworth, Chap 10, page 210.

The submission process

Email your submission in the first place in Word format to [email protected] clearly indicating the nature of the submission e.g. case series, research project.

Ensure that your copy includes information about the author (full name, qualifications, and email address).

Once your submission has been received you will receive feedback from the reviewers indicating whether the copy has been accepted for publication.


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There are a number of levels of acceptance:

□ acceptance: no amendments required;

□ acceptance: minor amendments required;

□ acceptance major amendments required;

□ rejection: not suitable for publication in the APCP Journal.


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NOTES

June 2018

www.apcp.org.uk

ASSOCIATION OF PAEDIATRICCHARTERED PHYSIOTHERAPISTS

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