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Hematology

ISSN (Print) 1607-8454 (Online) Journal homepage httpswwwtandfonlinecomloiyhem20

Modifying factors of the health belief modelassociated with missed clinic appointmentsamong individuals with sickle cell disease

Robert M Cronin Jane S Hankins Jeannie Byrd Brandi M Pernell AdetolaKassim Patricia Adams-Graves Alexis A Thompson Karen Kalinyak MichaelR DeBaun amp Marsha Treadwell

To cite this article Robert M Cronin Jane S Hankins Jeannie Byrd Brandi M Pernell AdetolaKassim Patricia Adams-Graves Alexis A Thompson Karen Kalinyak Michael R DeBaun ampMarsha Treadwell (2018) Modifying factors of the health belief model associated with missedclinic appointments among individuals with sickle cell disease Hematology 239 683-691 DOI1010801024533220181457200

To link to this article httpsdoiorg1010801024533220181457200

Published online 29 Mar 2018

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Modifying factors of the health belief model associated with missed clinicappointments among individuals with sickle cell diseaseRobert M Cronin abc Jane S Hankinsd Jeannie Byrde Brandi M Pernellef Adetola KassimgPatricia Adams-Gravesh Alexis A Thompsoni Karen Kalinyakj Michael R DeBaune and Marsha Treadwell k

aDepartment of Biomedical Informatics Vanderbilt University Medical Center Nashville TN USA bDepartment of Internal MedicineVanderbilt University Medical Center Nashville TN USA cDepartment of Pediatrics Vanderbilt University Medical Center Nashville TNUSA dDepartment of Hematology St Jude Childrenrsquos Research Hospital Memphis TN USA eDepartment of Pediatrics Division ofHematologyOncology Vanderbilt-Meharry Center for Excellence in Sickle Cell Disease Vanderbilt University Medical Center Nashville TNUSA fDepartment of Pediatrics Division of Hematology University of Alabama at Birmingham Birmingham AL USA gDepartment ofHematologyOncology Vanderbilt University Medical Center Nashville TN USA hDepartment of General Internal Medicine University ofTennessee Health Science Center Memphis TN USA iAnn and Robert H Lurie Childrenrsquos Hospital of Chicago Department of PediatricsNorthwestern University Chicago IL USA jDivision of Hematology in Cancer and Blood Diseases Institute University of CincinnatiCincinnati OH USA kDepartment of HematologyOncology UCSF Benioff Childrenrsquos Hospital Oakland Oakland CA USA

ABSTRACTObjectives Outpatient care is critical in the management of chronic diseases including sicklecell disease (SCD) Risk factors for poor adherence with clinic appointments in SCD are poorlydefined This exploratory study evaluated associations between modifying variables from theHealth Belief Model and missed appointmentsMethods We surveyed adults with SCD (n = 211) and caregivers of children with SCD (n = 331)between October 2014 and March 2016 in six centres across the US The survey tool utilized theframework of the Health Belief Model and included social determinants psychosocial variablessocial support health literacy and spiritualityResults A majority of adults (87) and caregivers of children (65) reported they missed aclinic appointment Children (as reported by caregivers) were less likely to missappointments than adults (OR022 95 CI(013039)) In adults financial insecurity (OR44995 CI(120 207)) health literacy (OR464 95 CI(133 1615)) and age (OR095 95 CI(091099)) were significantly associated with missed appointments In all participants lowerspirituality was associated with missed appointments (OR183 95CI(113 294)) The mostcommon reason for missing an appointment was forgetfulness (adults 31 children 26)A majority thought reminders would help (adults 83 children 71) using phone calls(adults 62 children 61) or text messages (adults 56 children 51)Conclusions Our findings demonstrate that modifying components of the Health Belief Modelincluding age financial security health literacy spirituality and lacking cues to action likereminders are important in missed appointments and addressing these factors couldimprove appointment-keeping for adults and children with SCD

KEYWORDSMissed clinic appointmentsvulnerable populationshealth care surveys sickle celldisease determinants ofhealth health belief model

Introduction

Sickle cell disease (SCD) is an inherited disorder ofhemoglobin that affects approximately 100000 indi-viduals in the US many of whom are African Americanand many of whom also live in poverty [1ndash4] Preven-tive care is critical to reducing complications and main-taining health and quality of life for patients with SCD[5] National guidelines recommend routine follow-upappointments every 6 months and more frequentlyfor patients experiencing complications or receivingdisease modifying therapies (eg hydroxyurea orchronic blood transfusion therapy) [67]

Most of the literature about barriers to attendingclinic appointments in SCD concerns the pediatricpopulation with very little focus on adults Publishedadherence rates for routine clinic appointments inSCD range from 46 to 77 [89] Adolescents with

SCD and their caregivers have reported several barriersto clinic attendance including competing activitieshealth status (both feeling well and not well enoughto attend) poor patient-provider relationshipsadverse prior clinical experiences and forgetfulness[10] Caregivers have also reported existing barriers toTranscranial Doppler (TCD) ultrasonography screeningthat included limited finances lack of transportationand inconvenient clinic hours [11] These barriers con-trasted with the results from a retrospective medicalrecord review that showed that privately insuredpatients were three times more likely to be adherentto TCD screenings [12] Health system barriers canalso reduce attendance to health-maintenance visitsamong individuals with SCD such as difficulties withcontacting providers extended wait times and incon-venient clinic hours [13] Individuals with SCD residing

copy 2018 Informa UK Limited trading as Taylor amp Francis Group

CONTACT Robert M Cronin robertcroninvanderbiltedu 2525 West End Suite 1475 Nashville TN 37203 USA

HEMATOLOGY2018 VOL 23 NO 9 683ndash691httpsdoiorg1010801024533220181457200

in rural areas experience longer travel distances andlimited access to primary and comprehensive outpatientservices resulting in higher rates of healthcare utilization[1415] Missed health-maintenance visits are costly tothe healthcare system and contribute to increasedacute care utilization [1617] Better understanding oflocal barriers to care in adults will help determinefuture interventions and funding allocations for services

The present study surveys a geographically diversepopulation of individuals with SCD about factors thataffect their utilization of ambulatory services Thesesurveys were based on the Health Belief Model (HBM ndash[18]) to improve understanding of variables that influ-ence behaviours that may lead to missed appointmentsThe HBM posits that taking a lsquohealth actionrsquo such askeeping outpatient clinic appointments is a function ofperceptions of the seriousness of the disease and thatthere are benefits but limited barriers to the healthaction 19 Modifying variables within the HBM mayfacilitate or hinder positive health actions (Figure 1)Missing appointments has been associated with socialdeterminants of health within the HBM includingfemale gender race and lower socioeconomic statusin primary care and other diseases [20ndash23] Missedappointments have also been associated with socialsupport in SCD [910] Spirituality and depressive symp-toms have impacts in other self-care management likemedication adherence in HIV but have not beenexplored in relation to clinic attendance in SCD [24]We examined the association between modifying vari-ables in the HBM model including several self-reportmeasures and self-reported missed appointments forindividuals with SCD We explored associationsbetween modifying components of the HBM andmissed appointments among adults with SCD and care-givers of children We also explored reasons for missedappointments and how to decrease them

Methods

Setting

We surveyed a convenience sample of adults with SCD(age ge18 years) and caregivers of children with SCD

(patients age lt 18 years) between October 2014 andMarch 2016 Surveys were completed at sites in threedistinct geographical regions the Midwest (CincinnatiChildrenrsquos Hospital Medical Center and Ann andRobert H Lurie Childrenrsquos Hospital of Chicago) theMid-South (University of Tennessee Health ScienceCenter St Jude Childrenrsquos Research Hospital and Van-derbilt University Medical Center) and the West (UCSFBenioff Childrenrsquos Hospital Oakland) Only individualswho could speak and read English were included Par-ticipants completed the survey only once

The Mid-South Clinical Data Research Network(CDRN) [25] was established in 2014 with fundingfrom the Patient-Centered Outcomes Research Insti-tute (PCORI) The 11 CDRN sites in the US have the fol-lowing collective goals to engage at minimum 11million patients across multiple healthcare systemsbuild infrastructure to share data and build novel infor-matics tools and perform comparative effectivenessresearch and pragmatic clinical trials The Mid-SouthCDRN survey tool was designed to obtain uniforminformation across obesity coronary heart diseaseand SCD cohorts The Institutional Review Boards ofthe participating sites approved all study proceduresand informed consent was obtained from allparticipants

Procedure

Individuals with SCD and their caregivers wererecruited using flyers placed in clinics and were intro-duced to the study by their clinicians during regularlyscheduled visit Procedures were described in moredetail by research staff during the informed consentsession The surveys were administered via computertablet but if a tablet was not available by paper-and-pencil Participants completed the surveys indepen-dently with a member of the research team nearbyto answer questions or provide assistance as neededParticipants received a gift card upon completion ofthe survey

Survey tool

The Mid-South CDRN researchers and key stakeholdersincluding healthcare providers psychologists socialworkers and individuals with SCD designed thesurvey tool based on selected components of theHBM framework including the following domainssocial determinants of health depressive symptomssocial support health literacy and spirituality (Figure1) as potentially influencing healthcare utilization andself-care in SCD [26] The complete survey consistedof 80 questions and took approximately 30 minutesto complete The reading level of the questions andsurvey tools ranged from 5th to 7th grade Outcomemeasures included self-reports of whether or not the

Figure 1 Health belief model for missed appointments Riskfactors evaluated are italicized

684 R M CRONIN ET AL

individual with SCD missed any appointments withinthe past year

Social determinants of healthThe Mid-South CDRN survey tool gathered socialdeterminants of health including age sex raceethnicity educational attainment difficulty payingmonthly bills and marital status Educational attain-ment difficulty paying bills and marital status werequestions asked about caregivers of children theremaining questions were asked about the childwith SCD We combined some categories of surveyresponses for ease of interpretation within theregression analyses The five levels of educationranging from some high school to post-graduatewere dichotomized into lsquoHigh school graduate orlessrsquo and lsquoSome college or morersquo Participants whoindicated that they were currently married or livingwith a stable partner were categorized as lsquoMarriedor living togetherrsquo while those who were singlewidowed divorced or separated were categorizedas lsquoUnmarriedrsquo The items inquiring about financialstatus lsquohow difficult is it for you (your family) to payyour monthly billsrsquo were compressed into lsquoNot veryor Not at all difficultrsquo versus lsquoVery or Somewhat diffi-cultrsquo These social determinants of health are modify-ing variables in the HBM

Depressive symptomsThe survey tool provided for evaluation of depressivesymptoms using the Patient Health Questionnaire(PHQ-2 [27]) a validated two-item screening for the fre-quency of depressed mood and anhedonia over thepast two weeks PHQ-2 scores range from 0 (not atall) to 6 (nearly every day) with a score of 3 suggestingthe need for further evaluation of depressive disorder[27] Caregivers were asked to assess their childrsquosdepressive symptoms Depressive symptoms are apsychological modifying variable in the HBM

Social supportParticipants rated their social supports using theENRICHD (Enhancing Recovery in Coronary HeartDisease) Social Support Inventory (ESSI [28]) Theyrated whether they had someone to whom theyfelt close who could give them advice show loveand affection and provide emotional support at dif-ficult times on a scale from None of the time (1) toAll of the time (5) so that higher scores indicatebetter access to social support Low support inthe ENRICHD has been defined as 2 or moreitems le2 or 2 or more items le3 and an adjustedoverall score le18 [29] Caregivers were asked toassess the social support of their child with SCDSocial support is a potential cue to action in theHBM

Health literacyHealth literacy or the ability to obtain read under-stand and use healthcare information to make appro-priate health decisions is an important component ofthe HBM [30] Health literacy was evaluated using theBrief Health Literacy Screening three items rated on afive-point scale indicating confidence in completingmedical forms without assistance need to ask forhelp in reading health-related materials and problemswith learning about SCD due to difficulty understand-ing written information Inadequate health literacycan be determined from one or a combination of allthree of these questions [3132] Responses of lsquosome-whatrsquo or better for the question lsquoHow confident areyou filling out medical forms by yourselfrsquo has beenused to define lsquogoodrsquo health literacy [31] Caregiverswere asked to assess their own health literacy Healthliteracy is a modifying variable between educationand ultimate health behaviours within the HBM

SpiritualityAn emerging focus of study within the HBM is on spiri-tuality as a barrier or resource for positive health action[33] particularly for African Americans [34] Participantsrated how spiritual they considered themselves to beusing a single item lsquohow spiritual or religious do youconsider yourself (or your child) to bersquo from very (1)to not at all (4) Based on the distribution of theresponses and for ease of analysis we dichotomizedthe variable into lsquoveryrsquo spiritual (option 1) and lsquonotveryrsquo spiritual (option 234) Caregivers were askedabout the spirituality of their child with SCD

Missed appointmentsAdults with SCD reported on whether they had missedan appointment within the previous twelve months byselecting from a potential list of contributing factors formissed appointments Caregivers reported on whethertheir child with SCD missed an appointment within theprevious twelve months in the same manner TheCDRN survey also asked what cues patients and care-givers preferred as reminders about appointments(eg text messages telephone calls)

Statistical analysis

Study data were collected de-identified and managedusing REDCap electronic data capture tools hosted atVanderbilt University [35] Data were entered eitherdirectly into the database as participants completedthem on computer tablets or transcribed from paper-based surveys Surveys were excluded if they weremissing information on age site or sex We useddescriptive statistics to summarize the social determi-nants of health and responses to questions in theremaining surveys Means and inter-quartile ranges

HEMATOLOGY 685

were used to describe continuous variables and pro-portions were used for categorical variables We alsorecorded the percent of missing responses for eachsurvey item

We created logistic regression models for theoutcome measure of missing appointments using vari-ables from the HBM ie social determinants of health(sex age ability to pay bills) depressive symptomshealth literacy spirituality and social support Logisticalregression models were based on the constructs of theHBM that were available We initially created a modelfor all participants but given that adults and childrenwith SCD have important differences in health carewe created a variable that dichotomized adults andchildren When we saw statistically significant differ-ences in our regression for all adults compared withchildren we created two new models for adults andchildren separately to further evaluate these differ-ences Analyses were performed in R version 322and p-values were considered significant if lt 005 [36]

Results

Demographics

A total of 573 individuals with SCD (adults and care-givers of children with SCD) completed the surveysAfter excluding surveys missed age sex or site ourfinal sample for analysis included 211 adults with SCDand 331 caregivers of children with SCD (n = 542)Table 1 shows distribution by sites of adults and pedi-atric patients

Modifying variables in the health belief modelvary among adults with SCD and children withSCD (as reported by their caregivers)

The most common education level for both adults withSCD and caregivers of children with SCD was lsquosomecollege educationrsquo Forty-five percent of the totalsample reported it was lsquosomewhatrsquo to lsquovery difficultrsquoto pay monthly bills Over 76 of the total samplerated themselves as lsquofairlyrsquo to lsquoveryrsquo spiritual or religiousThe mean score on the PHQ-2 for depression in adults(146 plusmn 155) was higher than what caregivers reportedfor children (084 plusmn 126) but both below the cut-off fordepression screening of 30 However 49 (232) adultsand 47 (142) children (as reported by their care-givers) had scores of 3 and above (Table 2) In ourpopulation 18 of all individuals with SCD evidencedmoderate to severe depressive symptoms based onthe PHQ-2 scores (adults 23 children (reported bycaregivers) 14) These numbers are slightly lowercompared with other studies in SCD where rates ofmoderate to severe depressive symptoms haveranged from 26 to 57 [37ndash39] Differences in thedepression screening tool used could likely account

for this difference The majority of adults and children(as reported by their caregivers) rated social supportand health literacy as lsquogoodrsquo (85 and 749respectively)

Forgetfulness was the most common reason formissed appointments and participants thoughta reminder would help them best

A majority of children (reported by caregivers 65)and adults (87) missed an appointment over thepast year (Table 3) although most also reported thatthey called ahead The most common reason for chil-dren and adults missing an appointment was forget-ting about the appointment (adults 36 children(reported by caregivers) 26) The next mostcommon reason for adults was that the time did notwork for them (29) and for caregivers of childrenwas not having a ride to get to the appointment(23) The most common reason for not calling whenthey missed an appointment was forgetting to call(58) A majority of participants thought a reminderwould help them make sure they got to clinic appoint-ments (75) Over half wanted a text message (53) ora phone call (61) Most people wanted a reminder theday of or day before the appointment (41)

Missed appointments were associated with agefinancial security spirituality and healthliteracy

Children were less likely to miss appointments thanadults (Odds Ratio (OR) 022 95 Confidence Interval(CI) = [010 051]) (Table 4) For the full sample difficultypaying bills (OR = 170 95 CI = [104 280]) and lessreported spirituality (OR = 183 95 CI = [113 294])was associated with missing appointments Amongadults younger age was associated with missingappointments (OR = 095 95 CI = [091 099]) Foradults difficulty paying bills (OR = 499 95 CI =[120 207]) and higher literacy (OR = 464 95 CI =[133 162]) were associated with missing appoint-ments For children with SCD younger age was associ-ated with more missed appointments (OR = 094 95CI = [088100])

Discussion

Understanding factors that influence missed appoint-ments is important when considering effective strat-egies to improve the care of individuals with SCDOur manuscript is one of the first to describe associ-ations between components of the HBM and missedappointments among individuals with SCD In ourstudy we identified the reasons for missed appoint-ments at multiple sickle cell centres across the USWe found that some but not all modifiers within the

686 R M CRONIN ET AL

HBM that were available to us were associated withmissed appointments for individuals with SCD Chil-dren and adults with SCD differed with regard to modi-fiers of the HBM that were associated with theoutcome indicating potentially different targets forimproving clinic appointment keeping Participantsalso thought that having better cues to action likereminders for clinic appointments would improvetheir attendance Our findings are consistent with find-ings from other research that have examined barriersand facilitators to appointment keeping for adoles-cents with SCD and caregivers of children with SCD[910] however research concerning appointmentkeeping for adults with SCD is limited

The CDRN data collection allowed for the first timeexamination of factors associated with appointmentkeeping for adults with SCD across the US Consistentwith other research younger transition-age adults (18

to 25 years) were most likely to miss appointments[4041] The disconnect between pediatric and adultcare is a well-known gap [42] with evidence thatpatients are unprepared for differing expectations inthe adult medical world ie making appointmentswithout the assistance of family or staff Interestinglyyounger children had more missed appointmentsthan older children which may be due to caregiversof younger children needing to get to work so theymiss more appointments as the caregiver is respon-sible to get the appointment However once theyoung adult with SCD transitions to adult care theymiss more appointments because they are more ontheir own Further it was surprising that higherhealth literacy was associated with missed appoint-ments for adults in our study Upon closer examinationcommon reasons that individuals with high health lit-eracy missed appointments were because they didnot want to miss school or work and they did notknow or forgot they had an appointment Adults withhigher literacy may have more responsibilities (egschool and work) and if they are in better health andperceive that other aspects of their lives are moreimportant they may not think they needed to beseen Further study is needed on the role that health lit-eracy plays in appointment keeping and other positivehealth behaviours for patients with SCD

We included spirituality as a modifier of the HBM inthe present study In a recent review [43] the impor-tance of spiritualityreligiosity in relation to improvedcoping and decreased health care utilization forpatients with SCD was supported We found significant

Table 1 Socio-demographics for participants with sickle cell disease (N ndash 542)

Variable Adults (N = 211)Childrena

(N = 331)Combined(N = 542)

Age Years (SD Range) 270 (180ndash700) 100 (00ndash170) 150 (00ndash700)Sex Male 91 (431)b 161 (486) 252 (465)

Female 120 (569) 170 (514) 290 (535)Raceethnicity Black African American African

or Afro-Caribbean203 (962) 324 (979) 527 (972)

Hispanic Latino or Spanish origin 5 (24) 5 (15) 10 (18)Some other race or origin 10 (47) 9 (27) 19 (35)

Highest degree or level of school completed High school graduate or less 90 (426) 134 (405) 224 (413)Some college or beyond 116 (550) 126 (381) 242 (446)

Household size Median (range) 4 (1 ndash14) 3 (1ndash8) 4 (1ndash14)Marital status Marriedliving together 54 (256) 111 (335) 165 (304)

Unmarried 147 (697) 220 (665) 377 (696)Spiritualityreligiosity Very 75 (355) 151 (456) 226 (417)

Fairly 87 (412) 102 (308) 189 (349)Slightlynot at all 39 (185) 60 (181) 99 (183)

Difficulty paying monthly bills Not verynot at all 117 (554) 176 (532) 293 (541)SomewhatVery 93 (441) 154 (465) 247 (456)

Site Midwest regionCincinnati 11 (52) 40 (121) 51 (94)Chicago 17 (81) 84 (254) 101 (186)Western regionOakland 47 (223) 0 (0) 47 (87)Mid-South regionSt Jude 6 (28) 156 (471) 162 (299)UTHSCc 47 (223) 0 (0) 47 (87)Vanderbilt 83 (393) 51 (154) 134 (247)

aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesbPercentages may not add up to 100 because of missing datacUTHSC = University of Tennessee Health Science Center

Table 2 Scores on standardized measures for the participants(n = 542) with sickle cell disease

MeasureAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Patient healthquestionnaire(PHQ-2 meanSD)

146 (155) 084 (126) 108 (141)

ENRICHD socialsupportinstrument (ESSI)(n)

Poor 47 (223) 31 (94) 78 (144)Good 164 (777) 300 (906) 464 (856)

Brief healthliteracyscreening (n)

Poor 61 (289) 75 (227) 136 (251)Good 150 (711) 256 (773) 406 (749)

aCaregivers were asked to report for their children under 18 years

HEMATOLOGY 687

associations between increased reported spiritualityand missed appointments in all individuals with SCDand further exploration of strategies to explicitly tapinto this potentially important coping mechanism isneeded

Since missed appointments are costly and contrib-ute to increased acute care utilization [1617] strategiesto improve appointment show rates could decreaseadmissions and readmissions The most commonreason for missed appointments was forgetting con-sistent with previous literature [10] The HBMrsquos cuesto action could be helpful in improving adherencewith appointments (Figure 1) Three quarters of partici-pants thought that reminders would be of help to themin making it to appointments The most common waythey wanted reminders was through phone callsHowever for adults aged 18 to 30 years comparedwith older adults text message reminders were pre-ferred (text message 63 phone call 57) demon-strating that different age groups prefer differenttypes of reminders A recent systematic review andmeta-analysis looked at reminders for clinic appoint-ments [44] It was found that people receiving notifica-tions improved clinic attendance by 23 However itwas also found that multiple notifications were signifi-cantly more effective than single notifications andvoice notifications were more effective than text

notifications Therefore clinic reminders which arenot done consistently at all the sites in this studycould help improve attendance

Among the factors that influenced missed appoint-ments in adults financial insecurity seemed to haveplayed a major role in missing appointments as adultswith SCD who had difficulty paying bills were fivetimesmore likely tomiss an appointment Financial inse-curity would be consistent with our findings that partici-pants missed appointments because they couldnrsquot getto the appointment (transportation issues) didnrsquot havethe money or did not have health insurance or co-payfor the appointment Improving the ability to get toappointments and meet expenses associated withappointments could be an important focus in improvingclinic attendance These findingsmay also highlight cog-nitive challenges [45] and vulnerability of the SCD popu-lation There is recognition that cognitive impairment inadults with SCDmay contribute to the risk of unemploy-ment while depression has been correlated to thechronic unpredictable pain events and psychosocial dis-tress associated with SCD [46] Providersmust be vigilantthat individuals with SCD and cognitive impairmentandor those with lower income and depressive symp-toms may require more attention to increase outpatientand decrease acute healthcare utilization whereappropriate

Table 3 Responses to questions about appointment keeping (n = 542)

Question ResponseAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Have youyour child missed an appointment forany reason over the past year

Yes 183 (867) 216 (653) 399 (736)No 26 (123) 114 (344) 140 (258)

Reasons youyour child missed an appointment Forgot 65 (308) 56 (259) 121 (303)Time did not work 53 (251) 42 (194) 95 (238)No way to get to the appointment 44 (209) 50 (231) 94 (236)Health impacted ability to make the appointment 40 (190) 9 (42) 49 (123)Did not know about the appointment 39 (185) 22 (102) 61 (153)Did not have the money to get to the appointment 28 (133) 17 (79) 45 (113)Had other things that needed to be done 23 (109) 12 (56) 35 (88)Couldnrsquot get off work to come to the appointment 22 (104) 34 (157) 56 (14)Family emergency 18 (85) 38 (176) 56 (14)Feeling fine and didnrsquot need to come to the appointment 11 (52) 5 (23) 16 (4)Didnrsquot want to miss workschool 10 (47) 25 (116) 35 (88)Did not have health insurance or co-pay 10 (47) 3 (14) 13 (33)Had a negative experience with previous appointment orlengthy wait times

5 (24) 0 (0) 5 (13)

Missed appointment without calling ahead No 110 (521) 247 (746) 357 (659)Yes 99 (469) 83 (251) 182 (336)

Reasons for not calling Forgot 62 (294) 44 (444) 106 (582)Did not know about the appointment 21 (100) 21 (420) 42 (231)Could not get anyone on the phone 11 (52) 10 (161) 21 (115)Did not know what number to call 5 (24) 3 (273) 8 (44)

What would help to make sure you come toeach clinic visit

A reminder 174 (825) 232 (701) 406 (749)Keeping appointments on a schedule 47 (223) 121 (366) 168 (310)Scheduling only necessary appointments instead of routineappointments

24 (114) 26 (79) 50 (92)

An incentive 12 (57) 20 (60) 32 (59)Education about need for preventative appointments 8 (38) 12 (36) 20 (37)

Preference for being reminded aboutappointments

Text message 117 (555) 170 (514) 287 (530)Phone call 131 (621) 202 (610) 333 (614)E-mail 42 (199) 97 (293) 139 (256)Postal Mail 33 (156) 120 (363) 153 (282)

Best schedule for reminder Day of or before appointment 94 (445) 128 (387) 222 (410)5 to 7 days before appointment 65 (308) 108 (326) 173 (319)All of the above 46 (218) 94 (284) 140 (258)

aCaregivers were asked to report for their children under 18 years

688 R M CRONIN ET AL

Several limitations in interpreting these results arenoted First findings are based on survey self-reportswhich can lead to recall bias Although the majorityof adults with SCD and children as reported by theircaregivers had missed an appointment informationabout missing appointments was based on self-reportonly Future studies should validate self-reports aboutmissing appointments with the electronic healthrecord Second while we were able to include respon-dents from three regions of the US in the surveyresponses may differ from other regions Third thiswas a convenience sample of participants who pre-sented to clinical sites providing SCD care While theirresponses may or may not be representative of thebroader population of patients with SCD and theirfamilies the clinical sites themselves did range fromacademic medical centres to community clinicsFourth spirituality was measured with a single-itemAlthough the item is part of a validated instrument[47] future studies with a complete spirituality scalecould be used to further evaluate the relationbetween spirituality and missing appointments Fifthwe do not know if the caregiver may have answeredsurvey questions about themselves in response to theprompt youyour child However coordinators whoadministered the surveys did not report that caregiversexpressed confusion with the survey instructions in this

regard Finally we were limited in what variables wecould evaluate in the present study because we wereusing an existing survey Insurance coverage anotherpotentially important variable in health care utilizationwas not captured in these surveys Also statisticallysome confidence intervals in the regression modelswere very wide suggesting a great deal of variabilitywithin the sample on some variables Direct evaluationof other components of the HBM and potentially otherimportant characteristics of participants was not poss-ible Further research is needed on the impact of rel-evant variables such as cognitive impairment andaccess to quality care on missed appointments forpopulations of individuals with SCD and their families

Conclusions

Our results demonstrated that modifiers of the HBMinfluence missed clinic appointments among individ-uals with SCD across the US Our findings supportthe importance of understanding social determinantsof health and other variables from the HBM inkeeping clinic appointments and highlight strategiesindividuals with SCD believed would help them withkeeping clinic appointments Among strategies remin-ders that are personalized to voice or text messagesbased on preferences improving the ability to get to

Table 4 Logistic regression model for missed appointment with variables from the health belief modelCombined model (N = 542)

Variable Odds ratio 95 CI Pr(gt|z|)

(Intercept) 769 (265223) lt0001Age group Children (lt18 yo)a 022 (013039) lt0001Sex Female 084 (052135) 0467Education Some college or more 063 (038104) 0072dagger

PHQ score 099 (082119) 0912Ability to pay bills Very or somewhat difficult 170 (104280) 0035Literacy High 139 (076255) 0284Spirituality Not very spiritual 183 (113294) 0013Social Support High 078 (034176) 055Adult model (N = 211)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 307 (222426) 0011Age 095 (091099) 0008Sex Female 045 (015139) 0168Education Some college or more 038 (012124) 011PHQ score 109 (076157) 064Ability to pay bills Very or somewhat difficult 499 (120207) 0027Literacy High 464 (133162) 0016Spirituality Not very spiritual 142 (046431) 054Social support High 080 (014449) 0798Model for children (N = 331)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 377 (098145) 0053Age 094 (088100) 0035Sex Female 095 (055166) 087Education Some college or more 068 (038121) 0189PHQ score 095 (076119) 063Ability to pay bills Very or somewhat difficult 144 (082253) 0199Literacy High 099 (048207) 0986Spirituality Not very spiritual 167 (095291) 0073dagger

Social support High 098 (037259) 097aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesp lt 005 daggerp lt 01

HEMATOLOGY 689

and pay for appointments and appealing to a personrsquosspirituality may improve clinic attendance Our findingssuggest potential targets to improve appointmentshow rates but also suggest areas in need of furtherstudy

Acknowledgements

The authors thank the members of the Vanderbilt-MeharryCenter of Excellence in Sickle Cell Disease for their thoughtfuland helpful comments in reviewing the manuscript BerthaDavis for regulatory matters Brittany L Myers DNP RN forsupport with patient recruitment and Natasha Dean forassistance with preparation of this manuscript

Disclosure statement

No potential conflict of interest was reported by the authors

Funding

This work was supported by PCORI [grant number CDRN1501-26498]

ORCID

Robert M Cronin httporcidorg0000-0003-1916-6521Marsha Treadwell httporcidorg0000-0003-0521-1846

References

[1] Brousseau DC Panepinto JA Nimmer M et al Thenumber of people with sickle-cell disease in theUnited States national and state estimates Am JHematol 201085(1)77ndash78

[2] Mvundura M Amendah D Kavanagh PL et al Healthcare utilization and expenditures for privately and pub-licly insured children with sickle cell disease in theUnited States Pediatr Blood Cancer 200953(4)642ndash646

[3] Steiner CA Miller JL Sickle cell disease patients in UShospitals 2004 Rockville (MD) Agency for HealthcareResearch and Quality (US) 2006

[4] Yusuf HR Atrash HK Grosse SD et al Emergency depart-ment visits made by patients with sickle cell disease adescriptive study 1999ndash2007 Am J Prev Med 201038(4 Suppl)S536ndashS541 doi101016jamepre201001001PubMed PMID 20331955 PubMed Central PMCIDPMCPMC4521762

[5] Mehta SR Afenyi-Annan A Byrns PJ et al Opportunitiesto improve outcomes in sickle cell disease Am FamPhysician 2006 Jul 1574(2)303ndash310 PubMed PMID16883928

[6] Yawn BP Buchanan GR Afenyi-Annan AN et alManagement of sickle cell disease summary of the2014 evidence-based report by expert panel membersJAMA 2014312(10)1033ndash1048 doi101001jama201410517 PubMed PMID 25203083

[7] Ware RE How I use hydroxyurea to treat young patientswith sickle cell anemia Blood 2010115(26)5300ndash5311doi101182blood-2009-04-146852 PubMed PMID20223921 PubMed Central PMCID PMCPMC2902131

[8] Thornburg CD Calatroni A Telen M et al Adherence tohydroxyurea therapy in children with sickle cell anemiaJ Pediatr 2010156(3)415ndash419 doi101016jjpeds200909044 PubMed PMID 19880135 PubMed CentralPMCID PMCPMC3901082

[9] Modi AC Crosby LE Hines J et al Feasibility of web-based technology to assess adherence to clinic appoint-ments in youth with sickle cell disease J PediatrHematol Oncol 201234(3)e93ndashe96 doi101097MPH0b013e318240d531 PubMed PMID 22278205PubMed Central PMCID PMCPMC3311776

[10] Crosby LE Modi AC Lemanek KL et al Perceived bar-riers to clinic appointments for adolescents with sicklecell disease J Pediatr Hematol Oncol 200931(8)571ndash576 doi101097MPH0b013e3181acd889 PubMedPMID 19636266 PubMed Central PMCIDPMCPMC2750821 eng

[11] Bollinger LM Nire KG Rhodes MM et al Caregiversrsquo per-spectives on barriers to transcranial Doppler screeningin children with sickle-cell disease Pediatr BloodCancer 201156(1)99ndash102 doi 101002pbc22780PubMed PMID 20842753

[12] Raphael JL Shetty PB Liu H et al A critical assessmentof transcranial Doppler screening rates in a large pedi-atric sickle cell center opportunities to improve health-care quality Pediatr Blood Cancer 200851(5)647ndash651doi101002pbc21677 PubMed PMID 18623200

[13] Jacob E Childress C Nathanson JD Barriers to care andquality of primary care services in children with sicklecell disease J Adv Nurs 201672(6)1417ndash1429 doi101111jan12756 PubMed PMID 26370255

[14] Schlenz AM Boan AD Lackland DT et al Needs assess-ment for patients with sickle cell disease in SouthCarolina 2012 Public Health Rep 2016131(1)108ndash116 doi101177003335491613100117 PubMed PMID26843676 PubMed Central PMCID PMCPMC4716478

[15] Brodsky MA Rodeghier M Sanger M et al Risk factorsfor 30-day readmission in adults with sickle-celldisease Am J Med 2017 doi101016jamjmed201612010 PubMed PMID 28065771

[16] Kheirkhah P Feng Q Travis LM et al Prevalence predic-tors and economic consequences of no-shows BMCHealth Serv Res 201516533 doi101186s12913-015-1243-z PubMed PMID 26769153 PubMed CentralPMCID PMCPMC4714455 eng

[17] Davies ML Goffman RM May JH et al Large-scale no-show patterns and distributions for clinic operationalresearch Healthcare (Basel) 20164(1) doi103390healthcare4010015 PubMed PMID 27417603 PubMedCentral PMCID PMCPMC4934549 eng

[18] Janz NK Becker MH The health belief model a decadelater Health Educ Behav 198411(1)1ndash47

[19] Stretcher VJ Rosenstock IM The health belief model InBaum A editor Cambridge handbook of psychologyhealth and medicine Cambridge CambridgeUniversity Press 1997 p 113ndash117 ISBN 0521430739

[20] Campbell B Staley D Matas M Who misses appoint-ments An empirical analysis Can J Psychiatry 199136(3)223ndash225 PubMed PMID 2059940

[21] Dove HG Schneider KC The usefulness of patientsrsquo indi-vidual characteristics in predicting no-shows in outpati-ent clinics Med Care 198119(7)734ndash740 PubMedPMID 7266121

[22] Goldman L Freidin R Cook EF et al A multivariateapproach to the prediction of no-show behavior in aprimary care center Arch Intern Med 1982142(3)563ndash567 PubMed PMID 7065791

690 R M CRONIN ET AL

individual with SCD missed any appointments withinthe past year

Social determinants of healthThe Mid-South CDRN survey tool gathered socialdeterminants of health including age sex raceethnicity educational attainment difficulty payingmonthly bills and marital status Educational attain-ment difficulty paying bills and marital status werequestions asked about caregivers of children theremaining questions were asked about the childwith SCD We combined some categories of surveyresponses for ease of interpretation within theregression analyses The five levels of educationranging from some high school to post-graduatewere dichotomized into lsquoHigh school graduate orlessrsquo and lsquoSome college or morersquo Participants whoindicated that they were currently married or livingwith a stable partner were categorized as lsquoMarriedor living togetherrsquo while those who were singlewidowed divorced or separated were categorizedas lsquoUnmarriedrsquo The items inquiring about financialstatus lsquohow difficult is it for you (your family) to payyour monthly billsrsquo were compressed into lsquoNot veryor Not at all difficultrsquo versus lsquoVery or Somewhat diffi-cultrsquo These social determinants of health are modify-ing variables in the HBM

Depressive symptomsThe survey tool provided for evaluation of depressivesymptoms using the Patient Health Questionnaire(PHQ-2 [27]) a validated two-item screening for the fre-quency of depressed mood and anhedonia over thepast two weeks PHQ-2 scores range from 0 (not atall) to 6 (nearly every day) with a score of 3 suggestingthe need for further evaluation of depressive disorder[27] Caregivers were asked to assess their childrsquosdepressive symptoms Depressive symptoms are apsychological modifying variable in the HBM

Social supportParticipants rated their social supports using theENRICHD (Enhancing Recovery in Coronary HeartDisease) Social Support Inventory (ESSI [28]) Theyrated whether they had someone to whom theyfelt close who could give them advice show loveand affection and provide emotional support at dif-ficult times on a scale from None of the time (1) toAll of the time (5) so that higher scores indicatebetter access to social support Low support inthe ENRICHD has been defined as 2 or moreitems le2 or 2 or more items le3 and an adjustedoverall score le18 [29] Caregivers were asked toassess the social support of their child with SCDSocial support is a potential cue to action in theHBM

Health literacyHealth literacy or the ability to obtain read under-stand and use healthcare information to make appro-priate health decisions is an important component ofthe HBM [30] Health literacy was evaluated using theBrief Health Literacy Screening three items rated on afive-point scale indicating confidence in completingmedical forms without assistance need to ask forhelp in reading health-related materials and problemswith learning about SCD due to difficulty understand-ing written information Inadequate health literacycan be determined from one or a combination of allthree of these questions [3132] Responses of lsquosome-whatrsquo or better for the question lsquoHow confident areyou filling out medical forms by yourselfrsquo has beenused to define lsquogoodrsquo health literacy [31] Caregiverswere asked to assess their own health literacy Healthliteracy is a modifying variable between educationand ultimate health behaviours within the HBM

SpiritualityAn emerging focus of study within the HBM is on spiri-tuality as a barrier or resource for positive health action[33] particularly for African Americans [34] Participantsrated how spiritual they considered themselves to beusing a single item lsquohow spiritual or religious do youconsider yourself (or your child) to bersquo from very (1)to not at all (4) Based on the distribution of theresponses and for ease of analysis we dichotomizedthe variable into lsquoveryrsquo spiritual (option 1) and lsquonotveryrsquo spiritual (option 234) Caregivers were askedabout the spirituality of their child with SCD

Missed appointmentsAdults with SCD reported on whether they had missedan appointment within the previous twelve months byselecting from a potential list of contributing factors formissed appointments Caregivers reported on whethertheir child with SCD missed an appointment within theprevious twelve months in the same manner TheCDRN survey also asked what cues patients and care-givers preferred as reminders about appointments(eg text messages telephone calls)

Statistical analysis

Study data were collected de-identified and managedusing REDCap electronic data capture tools hosted atVanderbilt University [35] Data were entered eitherdirectly into the database as participants completedthem on computer tablets or transcribed from paper-based surveys Surveys were excluded if they weremissing information on age site or sex We useddescriptive statistics to summarize the social determi-nants of health and responses to questions in theremaining surveys Means and inter-quartile ranges

HEMATOLOGY 685

were used to describe continuous variables and pro-portions were used for categorical variables We alsorecorded the percent of missing responses for eachsurvey item

We created logistic regression models for theoutcome measure of missing appointments using vari-ables from the HBM ie social determinants of health(sex age ability to pay bills) depressive symptomshealth literacy spirituality and social support Logisticalregression models were based on the constructs of theHBM that were available We initially created a modelfor all participants but given that adults and childrenwith SCD have important differences in health carewe created a variable that dichotomized adults andchildren When we saw statistically significant differ-ences in our regression for all adults compared withchildren we created two new models for adults andchildren separately to further evaluate these differ-ences Analyses were performed in R version 322and p-values were considered significant if lt 005 [36]

Results

Demographics

A total of 573 individuals with SCD (adults and care-givers of children with SCD) completed the surveysAfter excluding surveys missed age sex or site ourfinal sample for analysis included 211 adults with SCDand 331 caregivers of children with SCD (n = 542)Table 1 shows distribution by sites of adults and pedi-atric patients

Modifying variables in the health belief modelvary among adults with SCD and children withSCD (as reported by their caregivers)

The most common education level for both adults withSCD and caregivers of children with SCD was lsquosomecollege educationrsquo Forty-five percent of the totalsample reported it was lsquosomewhatrsquo to lsquovery difficultrsquoto pay monthly bills Over 76 of the total samplerated themselves as lsquofairlyrsquo to lsquoveryrsquo spiritual or religiousThe mean score on the PHQ-2 for depression in adults(146 plusmn 155) was higher than what caregivers reportedfor children (084 plusmn 126) but both below the cut-off fordepression screening of 30 However 49 (232) adultsand 47 (142) children (as reported by their care-givers) had scores of 3 and above (Table 2) In ourpopulation 18 of all individuals with SCD evidencedmoderate to severe depressive symptoms based onthe PHQ-2 scores (adults 23 children (reported bycaregivers) 14) These numbers are slightly lowercompared with other studies in SCD where rates ofmoderate to severe depressive symptoms haveranged from 26 to 57 [37ndash39] Differences in thedepression screening tool used could likely account

for this difference The majority of adults and children(as reported by their caregivers) rated social supportand health literacy as lsquogoodrsquo (85 and 749respectively)

Forgetfulness was the most common reason formissed appointments and participants thoughta reminder would help them best

A majority of children (reported by caregivers 65)and adults (87) missed an appointment over thepast year (Table 3) although most also reported thatthey called ahead The most common reason for chil-dren and adults missing an appointment was forget-ting about the appointment (adults 36 children(reported by caregivers) 26) The next mostcommon reason for adults was that the time did notwork for them (29) and for caregivers of childrenwas not having a ride to get to the appointment(23) The most common reason for not calling whenthey missed an appointment was forgetting to call(58) A majority of participants thought a reminderwould help them make sure they got to clinic appoint-ments (75) Over half wanted a text message (53) ora phone call (61) Most people wanted a reminder theday of or day before the appointment (41)

Missed appointments were associated with agefinancial security spirituality and healthliteracy

Children were less likely to miss appointments thanadults (Odds Ratio (OR) 022 95 Confidence Interval(CI) = [010 051]) (Table 4) For the full sample difficultypaying bills (OR = 170 95 CI = [104 280]) and lessreported spirituality (OR = 183 95 CI = [113 294])was associated with missing appointments Amongadults younger age was associated with missingappointments (OR = 095 95 CI = [091 099]) Foradults difficulty paying bills (OR = 499 95 CI =[120 207]) and higher literacy (OR = 464 95 CI =[133 162]) were associated with missing appoint-ments For children with SCD younger age was associ-ated with more missed appointments (OR = 094 95CI = [088100])

Discussion

Understanding factors that influence missed appoint-ments is important when considering effective strat-egies to improve the care of individuals with SCDOur manuscript is one of the first to describe associ-ations between components of the HBM and missedappointments among individuals with SCD In ourstudy we identified the reasons for missed appoint-ments at multiple sickle cell centres across the USWe found that some but not all modifiers within the

686 R M CRONIN ET AL

HBM that were available to us were associated withmissed appointments for individuals with SCD Chil-dren and adults with SCD differed with regard to modi-fiers of the HBM that were associated with theoutcome indicating potentially different targets forimproving clinic appointment keeping Participantsalso thought that having better cues to action likereminders for clinic appointments would improvetheir attendance Our findings are consistent with find-ings from other research that have examined barriersand facilitators to appointment keeping for adoles-cents with SCD and caregivers of children with SCD[910] however research concerning appointmentkeeping for adults with SCD is limited

The CDRN data collection allowed for the first timeexamination of factors associated with appointmentkeeping for adults with SCD across the US Consistentwith other research younger transition-age adults (18

to 25 years) were most likely to miss appointments[4041] The disconnect between pediatric and adultcare is a well-known gap [42] with evidence thatpatients are unprepared for differing expectations inthe adult medical world ie making appointmentswithout the assistance of family or staff Interestinglyyounger children had more missed appointmentsthan older children which may be due to caregiversof younger children needing to get to work so theymiss more appointments as the caregiver is respon-sible to get the appointment However once theyoung adult with SCD transitions to adult care theymiss more appointments because they are more ontheir own Further it was surprising that higherhealth literacy was associated with missed appoint-ments for adults in our study Upon closer examinationcommon reasons that individuals with high health lit-eracy missed appointments were because they didnot want to miss school or work and they did notknow or forgot they had an appointment Adults withhigher literacy may have more responsibilities (egschool and work) and if they are in better health andperceive that other aspects of their lives are moreimportant they may not think they needed to beseen Further study is needed on the role that health lit-eracy plays in appointment keeping and other positivehealth behaviours for patients with SCD

We included spirituality as a modifier of the HBM inthe present study In a recent review [43] the impor-tance of spiritualityreligiosity in relation to improvedcoping and decreased health care utilization forpatients with SCD was supported We found significant

Table 1 Socio-demographics for participants with sickle cell disease (N ndash 542)

Variable Adults (N = 211)Childrena

(N = 331)Combined(N = 542)

Age Years (SD Range) 270 (180ndash700) 100 (00ndash170) 150 (00ndash700)Sex Male 91 (431)b 161 (486) 252 (465)

Female 120 (569) 170 (514) 290 (535)Raceethnicity Black African American African

or Afro-Caribbean203 (962) 324 (979) 527 (972)

Hispanic Latino or Spanish origin 5 (24) 5 (15) 10 (18)Some other race or origin 10 (47) 9 (27) 19 (35)

Highest degree or level of school completed High school graduate or less 90 (426) 134 (405) 224 (413)Some college or beyond 116 (550) 126 (381) 242 (446)

Household size Median (range) 4 (1 ndash14) 3 (1ndash8) 4 (1ndash14)Marital status Marriedliving together 54 (256) 111 (335) 165 (304)

Unmarried 147 (697) 220 (665) 377 (696)Spiritualityreligiosity Very 75 (355) 151 (456) 226 (417)

Fairly 87 (412) 102 (308) 189 (349)Slightlynot at all 39 (185) 60 (181) 99 (183)

Difficulty paying monthly bills Not verynot at all 117 (554) 176 (532) 293 (541)SomewhatVery 93 (441) 154 (465) 247 (456)

Site Midwest regionCincinnati 11 (52) 40 (121) 51 (94)Chicago 17 (81) 84 (254) 101 (186)Western regionOakland 47 (223) 0 (0) 47 (87)Mid-South regionSt Jude 6 (28) 156 (471) 162 (299)UTHSCc 47 (223) 0 (0) 47 (87)Vanderbilt 83 (393) 51 (154) 134 (247)

aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesbPercentages may not add up to 100 because of missing datacUTHSC = University of Tennessee Health Science Center

Table 2 Scores on standardized measures for the participants(n = 542) with sickle cell disease

MeasureAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Patient healthquestionnaire(PHQ-2 meanSD)

146 (155) 084 (126) 108 (141)

ENRICHD socialsupportinstrument (ESSI)(n)

Poor 47 (223) 31 (94) 78 (144)Good 164 (777) 300 (906) 464 (856)

Brief healthliteracyscreening (n)

Poor 61 (289) 75 (227) 136 (251)Good 150 (711) 256 (773) 406 (749)

aCaregivers were asked to report for their children under 18 years

HEMATOLOGY 687

associations between increased reported spiritualityand missed appointments in all individuals with SCDand further exploration of strategies to explicitly tapinto this potentially important coping mechanism isneeded

Since missed appointments are costly and contrib-ute to increased acute care utilization [1617] strategiesto improve appointment show rates could decreaseadmissions and readmissions The most commonreason for missed appointments was forgetting con-sistent with previous literature [10] The HBMrsquos cuesto action could be helpful in improving adherencewith appointments (Figure 1) Three quarters of partici-pants thought that reminders would be of help to themin making it to appointments The most common waythey wanted reminders was through phone callsHowever for adults aged 18 to 30 years comparedwith older adults text message reminders were pre-ferred (text message 63 phone call 57) demon-strating that different age groups prefer differenttypes of reminders A recent systematic review andmeta-analysis looked at reminders for clinic appoint-ments [44] It was found that people receiving notifica-tions improved clinic attendance by 23 However itwas also found that multiple notifications were signifi-cantly more effective than single notifications andvoice notifications were more effective than text

notifications Therefore clinic reminders which arenot done consistently at all the sites in this studycould help improve attendance

Among the factors that influenced missed appoint-ments in adults financial insecurity seemed to haveplayed a major role in missing appointments as adultswith SCD who had difficulty paying bills were fivetimesmore likely tomiss an appointment Financial inse-curity would be consistent with our findings that partici-pants missed appointments because they couldnrsquot getto the appointment (transportation issues) didnrsquot havethe money or did not have health insurance or co-payfor the appointment Improving the ability to get toappointments and meet expenses associated withappointments could be an important focus in improvingclinic attendance These findingsmay also highlight cog-nitive challenges [45] and vulnerability of the SCD popu-lation There is recognition that cognitive impairment inadults with SCDmay contribute to the risk of unemploy-ment while depression has been correlated to thechronic unpredictable pain events and psychosocial dis-tress associated with SCD [46] Providersmust be vigilantthat individuals with SCD and cognitive impairmentandor those with lower income and depressive symp-toms may require more attention to increase outpatientand decrease acute healthcare utilization whereappropriate

Table 3 Responses to questions about appointment keeping (n = 542)

Question ResponseAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Have youyour child missed an appointment forany reason over the past year

Yes 183 (867) 216 (653) 399 (736)No 26 (123) 114 (344) 140 (258)

Reasons youyour child missed an appointment Forgot 65 (308) 56 (259) 121 (303)Time did not work 53 (251) 42 (194) 95 (238)No way to get to the appointment 44 (209) 50 (231) 94 (236)Health impacted ability to make the appointment 40 (190) 9 (42) 49 (123)Did not know about the appointment 39 (185) 22 (102) 61 (153)Did not have the money to get to the appointment 28 (133) 17 (79) 45 (113)Had other things that needed to be done 23 (109) 12 (56) 35 (88)Couldnrsquot get off work to come to the appointment 22 (104) 34 (157) 56 (14)Family emergency 18 (85) 38 (176) 56 (14)Feeling fine and didnrsquot need to come to the appointment 11 (52) 5 (23) 16 (4)Didnrsquot want to miss workschool 10 (47) 25 (116) 35 (88)Did not have health insurance or co-pay 10 (47) 3 (14) 13 (33)Had a negative experience with previous appointment orlengthy wait times

5 (24) 0 (0) 5 (13)

Missed appointment without calling ahead No 110 (521) 247 (746) 357 (659)Yes 99 (469) 83 (251) 182 (336)

Reasons for not calling Forgot 62 (294) 44 (444) 106 (582)Did not know about the appointment 21 (100) 21 (420) 42 (231)Could not get anyone on the phone 11 (52) 10 (161) 21 (115)Did not know what number to call 5 (24) 3 (273) 8 (44)

What would help to make sure you come toeach clinic visit

A reminder 174 (825) 232 (701) 406 (749)Keeping appointments on a schedule 47 (223) 121 (366) 168 (310)Scheduling only necessary appointments instead of routineappointments

24 (114) 26 (79) 50 (92)

An incentive 12 (57) 20 (60) 32 (59)Education about need for preventative appointments 8 (38) 12 (36) 20 (37)

Preference for being reminded aboutappointments

Text message 117 (555) 170 (514) 287 (530)Phone call 131 (621) 202 (610) 333 (614)E-mail 42 (199) 97 (293) 139 (256)Postal Mail 33 (156) 120 (363) 153 (282)

Best schedule for reminder Day of or before appointment 94 (445) 128 (387) 222 (410)5 to 7 days before appointment 65 (308) 108 (326) 173 (319)All of the above 46 (218) 94 (284) 140 (258)

aCaregivers were asked to report for their children under 18 years

688 R M CRONIN ET AL

Several limitations in interpreting these results arenoted First findings are based on survey self-reportswhich can lead to recall bias Although the majorityof adults with SCD and children as reported by theircaregivers had missed an appointment informationabout missing appointments was based on self-reportonly Future studies should validate self-reports aboutmissing appointments with the electronic healthrecord Second while we were able to include respon-dents from three regions of the US in the surveyresponses may differ from other regions Third thiswas a convenience sample of participants who pre-sented to clinical sites providing SCD care While theirresponses may or may not be representative of thebroader population of patients with SCD and theirfamilies the clinical sites themselves did range fromacademic medical centres to community clinicsFourth spirituality was measured with a single-itemAlthough the item is part of a validated instrument[47] future studies with a complete spirituality scalecould be used to further evaluate the relationbetween spirituality and missing appointments Fifthwe do not know if the caregiver may have answeredsurvey questions about themselves in response to theprompt youyour child However coordinators whoadministered the surveys did not report that caregiversexpressed confusion with the survey instructions in this

regard Finally we were limited in what variables wecould evaluate in the present study because we wereusing an existing survey Insurance coverage anotherpotentially important variable in health care utilizationwas not captured in these surveys Also statisticallysome confidence intervals in the regression modelswere very wide suggesting a great deal of variabilitywithin the sample on some variables Direct evaluationof other components of the HBM and potentially otherimportant characteristics of participants was not poss-ible Further research is needed on the impact of rel-evant variables such as cognitive impairment andaccess to quality care on missed appointments forpopulations of individuals with SCD and their families

Conclusions

Our results demonstrated that modifiers of the HBMinfluence missed clinic appointments among individ-uals with SCD across the US Our findings supportthe importance of understanding social determinantsof health and other variables from the HBM inkeeping clinic appointments and highlight strategiesindividuals with SCD believed would help them withkeeping clinic appointments Among strategies remin-ders that are personalized to voice or text messagesbased on preferences improving the ability to get to

Table 4 Logistic regression model for missed appointment with variables from the health belief modelCombined model (N = 542)

Variable Odds ratio 95 CI Pr(gt|z|)

(Intercept) 769 (265223) lt0001Age group Children (lt18 yo)a 022 (013039) lt0001Sex Female 084 (052135) 0467Education Some college or more 063 (038104) 0072dagger

PHQ score 099 (082119) 0912Ability to pay bills Very or somewhat difficult 170 (104280) 0035Literacy High 139 (076255) 0284Spirituality Not very spiritual 183 (113294) 0013Social Support High 078 (034176) 055Adult model (N = 211)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 307 (222426) 0011Age 095 (091099) 0008Sex Female 045 (015139) 0168Education Some college or more 038 (012124) 011PHQ score 109 (076157) 064Ability to pay bills Very or somewhat difficult 499 (120207) 0027Literacy High 464 (133162) 0016Spirituality Not very spiritual 142 (046431) 054Social support High 080 (014449) 0798Model for children (N = 331)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 377 (098145) 0053Age 094 (088100) 0035Sex Female 095 (055166) 087Education Some college or more 068 (038121) 0189PHQ score 095 (076119) 063Ability to pay bills Very or somewhat difficult 144 (082253) 0199Literacy High 099 (048207) 0986Spirituality Not very spiritual 167 (095291) 0073dagger

Social support High 098 (037259) 097aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesp lt 005 daggerp lt 01

HEMATOLOGY 689

and pay for appointments and appealing to a personrsquosspirituality may improve clinic attendance Our findingssuggest potential targets to improve appointmentshow rates but also suggest areas in need of furtherstudy

Acknowledgements

The authors thank the members of the Vanderbilt-MeharryCenter of Excellence in Sickle Cell Disease for their thoughtfuland helpful comments in reviewing the manuscript BerthaDavis for regulatory matters Brittany L Myers DNP RN forsupport with patient recruitment and Natasha Dean forassistance with preparation of this manuscript

Disclosure statement

No potential conflict of interest was reported by the authors

Funding

This work was supported by PCORI [grant number CDRN1501-26498]

ORCID

Robert M Cronin httporcidorg0000-0003-1916-6521Marsha Treadwell httporcidorg0000-0003-0521-1846

References

[1] Brousseau DC Panepinto JA Nimmer M et al Thenumber of people with sickle-cell disease in theUnited States national and state estimates Am JHematol 201085(1)77ndash78

[2] Mvundura M Amendah D Kavanagh PL et al Healthcare utilization and expenditures for privately and pub-licly insured children with sickle cell disease in theUnited States Pediatr Blood Cancer 200953(4)642ndash646

[3] Steiner CA Miller JL Sickle cell disease patients in UShospitals 2004 Rockville (MD) Agency for HealthcareResearch and Quality (US) 2006

[4] Yusuf HR Atrash HK Grosse SD et al Emergency depart-ment visits made by patients with sickle cell disease adescriptive study 1999ndash2007 Am J Prev Med 201038(4 Suppl)S536ndashS541 doi101016jamepre201001001PubMed PMID 20331955 PubMed Central PMCIDPMCPMC4521762

[5] Mehta SR Afenyi-Annan A Byrns PJ et al Opportunitiesto improve outcomes in sickle cell disease Am FamPhysician 2006 Jul 1574(2)303ndash310 PubMed PMID16883928

[6] Yawn BP Buchanan GR Afenyi-Annan AN et alManagement of sickle cell disease summary of the2014 evidence-based report by expert panel membersJAMA 2014312(10)1033ndash1048 doi101001jama201410517 PubMed PMID 25203083

[7] Ware RE How I use hydroxyurea to treat young patientswith sickle cell anemia Blood 2010115(26)5300ndash5311doi101182blood-2009-04-146852 PubMed PMID20223921 PubMed Central PMCID PMCPMC2902131

[8] Thornburg CD Calatroni A Telen M et al Adherence tohydroxyurea therapy in children with sickle cell anemiaJ Pediatr 2010156(3)415ndash419 doi101016jjpeds200909044 PubMed PMID 19880135 PubMed CentralPMCID PMCPMC3901082

[9] Modi AC Crosby LE Hines J et al Feasibility of web-based technology to assess adherence to clinic appoint-ments in youth with sickle cell disease J PediatrHematol Oncol 201234(3)e93ndashe96 doi101097MPH0b013e318240d531 PubMed PMID 22278205PubMed Central PMCID PMCPMC3311776

[10] Crosby LE Modi AC Lemanek KL et al Perceived bar-riers to clinic appointments for adolescents with sicklecell disease J Pediatr Hematol Oncol 200931(8)571ndash576 doi101097MPH0b013e3181acd889 PubMedPMID 19636266 PubMed Central PMCIDPMCPMC2750821 eng

[11] Bollinger LM Nire KG Rhodes MM et al Caregiversrsquo per-spectives on barriers to transcranial Doppler screeningin children with sickle-cell disease Pediatr BloodCancer 201156(1)99ndash102 doi 101002pbc22780PubMed PMID 20842753

[12] Raphael JL Shetty PB Liu H et al A critical assessmentof transcranial Doppler screening rates in a large pedi-atric sickle cell center opportunities to improve health-care quality Pediatr Blood Cancer 200851(5)647ndash651doi101002pbc21677 PubMed PMID 18623200

[13] Jacob E Childress C Nathanson JD Barriers to care andquality of primary care services in children with sicklecell disease J Adv Nurs 201672(6)1417ndash1429 doi101111jan12756 PubMed PMID 26370255

[14] Schlenz AM Boan AD Lackland DT et al Needs assess-ment for patients with sickle cell disease in SouthCarolina 2012 Public Health Rep 2016131(1)108ndash116 doi101177003335491613100117 PubMed PMID26843676 PubMed Central PMCID PMCPMC4716478

[15] Brodsky MA Rodeghier M Sanger M et al Risk factorsfor 30-day readmission in adults with sickle-celldisease Am J Med 2017 doi101016jamjmed201612010 PubMed PMID 28065771

[16] Kheirkhah P Feng Q Travis LM et al Prevalence predic-tors and economic consequences of no-shows BMCHealth Serv Res 201516533 doi101186s12913-015-1243-z PubMed PMID 26769153 PubMed CentralPMCID PMCPMC4714455 eng

[17] Davies ML Goffman RM May JH et al Large-scale no-show patterns and distributions for clinic operationalresearch Healthcare (Basel) 20164(1) doi103390healthcare4010015 PubMed PMID 27417603 PubMedCentral PMCID PMCPMC4934549 eng

[18] Janz NK Becker MH The health belief model a decadelater Health Educ Behav 198411(1)1ndash47

[19] Stretcher VJ Rosenstock IM The health belief model InBaum A editor Cambridge handbook of psychologyhealth and medicine Cambridge CambridgeUniversity Press 1997 p 113ndash117 ISBN 0521430739

[20] Campbell B Staley D Matas M Who misses appoint-ments An empirical analysis Can J Psychiatry 199136(3)223ndash225 PubMed PMID 2059940

[21] Dove HG Schneider KC The usefulness of patientsrsquo indi-vidual characteristics in predicting no-shows in outpati-ent clinics Med Care 198119(7)734ndash740 PubMedPMID 7266121

[22] Goldman L Freidin R Cook EF et al A multivariateapproach to the prediction of no-show behavior in aprimary care center Arch Intern Med 1982142(3)563ndash567 PubMed PMID 7065791

690 R M CRONIN ET AL

were used to describe continuous variables and pro-portions were used for categorical variables We alsorecorded the percent of missing responses for eachsurvey item

We created logistic regression models for theoutcome measure of missing appointments using vari-ables from the HBM ie social determinants of health(sex age ability to pay bills) depressive symptomshealth literacy spirituality and social support Logisticalregression models were based on the constructs of theHBM that were available We initially created a modelfor all participants but given that adults and childrenwith SCD have important differences in health carewe created a variable that dichotomized adults andchildren When we saw statistically significant differ-ences in our regression for all adults compared withchildren we created two new models for adults andchildren separately to further evaluate these differ-ences Analyses were performed in R version 322and p-values were considered significant if lt 005 [36]

Results

Demographics

A total of 573 individuals with SCD (adults and care-givers of children with SCD) completed the surveysAfter excluding surveys missed age sex or site ourfinal sample for analysis included 211 adults with SCDand 331 caregivers of children with SCD (n = 542)Table 1 shows distribution by sites of adults and pedi-atric patients

Modifying variables in the health belief modelvary among adults with SCD and children withSCD (as reported by their caregivers)

The most common education level for both adults withSCD and caregivers of children with SCD was lsquosomecollege educationrsquo Forty-five percent of the totalsample reported it was lsquosomewhatrsquo to lsquovery difficultrsquoto pay monthly bills Over 76 of the total samplerated themselves as lsquofairlyrsquo to lsquoveryrsquo spiritual or religiousThe mean score on the PHQ-2 for depression in adults(146 plusmn 155) was higher than what caregivers reportedfor children (084 plusmn 126) but both below the cut-off fordepression screening of 30 However 49 (232) adultsand 47 (142) children (as reported by their care-givers) had scores of 3 and above (Table 2) In ourpopulation 18 of all individuals with SCD evidencedmoderate to severe depressive symptoms based onthe PHQ-2 scores (adults 23 children (reported bycaregivers) 14) These numbers are slightly lowercompared with other studies in SCD where rates ofmoderate to severe depressive symptoms haveranged from 26 to 57 [37ndash39] Differences in thedepression screening tool used could likely account

for this difference The majority of adults and children(as reported by their caregivers) rated social supportand health literacy as lsquogoodrsquo (85 and 749respectively)

Forgetfulness was the most common reason formissed appointments and participants thoughta reminder would help them best

A majority of children (reported by caregivers 65)and adults (87) missed an appointment over thepast year (Table 3) although most also reported thatthey called ahead The most common reason for chil-dren and adults missing an appointment was forget-ting about the appointment (adults 36 children(reported by caregivers) 26) The next mostcommon reason for adults was that the time did notwork for them (29) and for caregivers of childrenwas not having a ride to get to the appointment(23) The most common reason for not calling whenthey missed an appointment was forgetting to call(58) A majority of participants thought a reminderwould help them make sure they got to clinic appoint-ments (75) Over half wanted a text message (53) ora phone call (61) Most people wanted a reminder theday of or day before the appointment (41)

Missed appointments were associated with agefinancial security spirituality and healthliteracy

Children were less likely to miss appointments thanadults (Odds Ratio (OR) 022 95 Confidence Interval(CI) = [010 051]) (Table 4) For the full sample difficultypaying bills (OR = 170 95 CI = [104 280]) and lessreported spirituality (OR = 183 95 CI = [113 294])was associated with missing appointments Amongadults younger age was associated with missingappointments (OR = 095 95 CI = [091 099]) Foradults difficulty paying bills (OR = 499 95 CI =[120 207]) and higher literacy (OR = 464 95 CI =[133 162]) were associated with missing appoint-ments For children with SCD younger age was associ-ated with more missed appointments (OR = 094 95CI = [088100])

Discussion

Understanding factors that influence missed appoint-ments is important when considering effective strat-egies to improve the care of individuals with SCDOur manuscript is one of the first to describe associ-ations between components of the HBM and missedappointments among individuals with SCD In ourstudy we identified the reasons for missed appoint-ments at multiple sickle cell centres across the USWe found that some but not all modifiers within the

686 R M CRONIN ET AL

HBM that were available to us were associated withmissed appointments for individuals with SCD Chil-dren and adults with SCD differed with regard to modi-fiers of the HBM that were associated with theoutcome indicating potentially different targets forimproving clinic appointment keeping Participantsalso thought that having better cues to action likereminders for clinic appointments would improvetheir attendance Our findings are consistent with find-ings from other research that have examined barriersand facilitators to appointment keeping for adoles-cents with SCD and caregivers of children with SCD[910] however research concerning appointmentkeeping for adults with SCD is limited

The CDRN data collection allowed for the first timeexamination of factors associated with appointmentkeeping for adults with SCD across the US Consistentwith other research younger transition-age adults (18

to 25 years) were most likely to miss appointments[4041] The disconnect between pediatric and adultcare is a well-known gap [42] with evidence thatpatients are unprepared for differing expectations inthe adult medical world ie making appointmentswithout the assistance of family or staff Interestinglyyounger children had more missed appointmentsthan older children which may be due to caregiversof younger children needing to get to work so theymiss more appointments as the caregiver is respon-sible to get the appointment However once theyoung adult with SCD transitions to adult care theymiss more appointments because they are more ontheir own Further it was surprising that higherhealth literacy was associated with missed appoint-ments for adults in our study Upon closer examinationcommon reasons that individuals with high health lit-eracy missed appointments were because they didnot want to miss school or work and they did notknow or forgot they had an appointment Adults withhigher literacy may have more responsibilities (egschool and work) and if they are in better health andperceive that other aspects of their lives are moreimportant they may not think they needed to beseen Further study is needed on the role that health lit-eracy plays in appointment keeping and other positivehealth behaviours for patients with SCD

We included spirituality as a modifier of the HBM inthe present study In a recent review [43] the impor-tance of spiritualityreligiosity in relation to improvedcoping and decreased health care utilization forpatients with SCD was supported We found significant

Table 1 Socio-demographics for participants with sickle cell disease (N ndash 542)

Variable Adults (N = 211)Childrena

(N = 331)Combined(N = 542)

Age Years (SD Range) 270 (180ndash700) 100 (00ndash170) 150 (00ndash700)Sex Male 91 (431)b 161 (486) 252 (465)

Female 120 (569) 170 (514) 290 (535)Raceethnicity Black African American African

or Afro-Caribbean203 (962) 324 (979) 527 (972)

Hispanic Latino or Spanish origin 5 (24) 5 (15) 10 (18)Some other race or origin 10 (47) 9 (27) 19 (35)

Highest degree or level of school completed High school graduate or less 90 (426) 134 (405) 224 (413)Some college or beyond 116 (550) 126 (381) 242 (446)

Household size Median (range) 4 (1 ndash14) 3 (1ndash8) 4 (1ndash14)Marital status Marriedliving together 54 (256) 111 (335) 165 (304)

Unmarried 147 (697) 220 (665) 377 (696)Spiritualityreligiosity Very 75 (355) 151 (456) 226 (417)

Fairly 87 (412) 102 (308) 189 (349)Slightlynot at all 39 (185) 60 (181) 99 (183)

Difficulty paying monthly bills Not verynot at all 117 (554) 176 (532) 293 (541)SomewhatVery 93 (441) 154 (465) 247 (456)

Site Midwest regionCincinnati 11 (52) 40 (121) 51 (94)Chicago 17 (81) 84 (254) 101 (186)Western regionOakland 47 (223) 0 (0) 47 (87)Mid-South regionSt Jude 6 (28) 156 (471) 162 (299)UTHSCc 47 (223) 0 (0) 47 (87)Vanderbilt 83 (393) 51 (154) 134 (247)

aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesbPercentages may not add up to 100 because of missing datacUTHSC = University of Tennessee Health Science Center

Table 2 Scores on standardized measures for the participants(n = 542) with sickle cell disease

MeasureAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Patient healthquestionnaire(PHQ-2 meanSD)

146 (155) 084 (126) 108 (141)

ENRICHD socialsupportinstrument (ESSI)(n)

Poor 47 (223) 31 (94) 78 (144)Good 164 (777) 300 (906) 464 (856)

Brief healthliteracyscreening (n)

Poor 61 (289) 75 (227) 136 (251)Good 150 (711) 256 (773) 406 (749)

aCaregivers were asked to report for their children under 18 years

HEMATOLOGY 687

associations between increased reported spiritualityand missed appointments in all individuals with SCDand further exploration of strategies to explicitly tapinto this potentially important coping mechanism isneeded

Since missed appointments are costly and contrib-ute to increased acute care utilization [1617] strategiesto improve appointment show rates could decreaseadmissions and readmissions The most commonreason for missed appointments was forgetting con-sistent with previous literature [10] The HBMrsquos cuesto action could be helpful in improving adherencewith appointments (Figure 1) Three quarters of partici-pants thought that reminders would be of help to themin making it to appointments The most common waythey wanted reminders was through phone callsHowever for adults aged 18 to 30 years comparedwith older adults text message reminders were pre-ferred (text message 63 phone call 57) demon-strating that different age groups prefer differenttypes of reminders A recent systematic review andmeta-analysis looked at reminders for clinic appoint-ments [44] It was found that people receiving notifica-tions improved clinic attendance by 23 However itwas also found that multiple notifications were signifi-cantly more effective than single notifications andvoice notifications were more effective than text

notifications Therefore clinic reminders which arenot done consistently at all the sites in this studycould help improve attendance

Among the factors that influenced missed appoint-ments in adults financial insecurity seemed to haveplayed a major role in missing appointments as adultswith SCD who had difficulty paying bills were fivetimesmore likely tomiss an appointment Financial inse-curity would be consistent with our findings that partici-pants missed appointments because they couldnrsquot getto the appointment (transportation issues) didnrsquot havethe money or did not have health insurance or co-payfor the appointment Improving the ability to get toappointments and meet expenses associated withappointments could be an important focus in improvingclinic attendance These findingsmay also highlight cog-nitive challenges [45] and vulnerability of the SCD popu-lation There is recognition that cognitive impairment inadults with SCDmay contribute to the risk of unemploy-ment while depression has been correlated to thechronic unpredictable pain events and psychosocial dis-tress associated with SCD [46] Providersmust be vigilantthat individuals with SCD and cognitive impairmentandor those with lower income and depressive symp-toms may require more attention to increase outpatientand decrease acute healthcare utilization whereappropriate

Table 3 Responses to questions about appointment keeping (n = 542)

Question ResponseAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Have youyour child missed an appointment forany reason over the past year

Yes 183 (867) 216 (653) 399 (736)No 26 (123) 114 (344) 140 (258)

Reasons youyour child missed an appointment Forgot 65 (308) 56 (259) 121 (303)Time did not work 53 (251) 42 (194) 95 (238)No way to get to the appointment 44 (209) 50 (231) 94 (236)Health impacted ability to make the appointment 40 (190) 9 (42) 49 (123)Did not know about the appointment 39 (185) 22 (102) 61 (153)Did not have the money to get to the appointment 28 (133) 17 (79) 45 (113)Had other things that needed to be done 23 (109) 12 (56) 35 (88)Couldnrsquot get off work to come to the appointment 22 (104) 34 (157) 56 (14)Family emergency 18 (85) 38 (176) 56 (14)Feeling fine and didnrsquot need to come to the appointment 11 (52) 5 (23) 16 (4)Didnrsquot want to miss workschool 10 (47) 25 (116) 35 (88)Did not have health insurance or co-pay 10 (47) 3 (14) 13 (33)Had a negative experience with previous appointment orlengthy wait times

5 (24) 0 (0) 5 (13)

Missed appointment without calling ahead No 110 (521) 247 (746) 357 (659)Yes 99 (469) 83 (251) 182 (336)

Reasons for not calling Forgot 62 (294) 44 (444) 106 (582)Did not know about the appointment 21 (100) 21 (420) 42 (231)Could not get anyone on the phone 11 (52) 10 (161) 21 (115)Did not know what number to call 5 (24) 3 (273) 8 (44)

What would help to make sure you come toeach clinic visit

A reminder 174 (825) 232 (701) 406 (749)Keeping appointments on a schedule 47 (223) 121 (366) 168 (310)Scheduling only necessary appointments instead of routineappointments

24 (114) 26 (79) 50 (92)

An incentive 12 (57) 20 (60) 32 (59)Education about need for preventative appointments 8 (38) 12 (36) 20 (37)

Preference for being reminded aboutappointments

Text message 117 (555) 170 (514) 287 (530)Phone call 131 (621) 202 (610) 333 (614)E-mail 42 (199) 97 (293) 139 (256)Postal Mail 33 (156) 120 (363) 153 (282)

Best schedule for reminder Day of or before appointment 94 (445) 128 (387) 222 (410)5 to 7 days before appointment 65 (308) 108 (326) 173 (319)All of the above 46 (218) 94 (284) 140 (258)

aCaregivers were asked to report for their children under 18 years

688 R M CRONIN ET AL

Several limitations in interpreting these results arenoted First findings are based on survey self-reportswhich can lead to recall bias Although the majorityof adults with SCD and children as reported by theircaregivers had missed an appointment informationabout missing appointments was based on self-reportonly Future studies should validate self-reports aboutmissing appointments with the electronic healthrecord Second while we were able to include respon-dents from three regions of the US in the surveyresponses may differ from other regions Third thiswas a convenience sample of participants who pre-sented to clinical sites providing SCD care While theirresponses may or may not be representative of thebroader population of patients with SCD and theirfamilies the clinical sites themselves did range fromacademic medical centres to community clinicsFourth spirituality was measured with a single-itemAlthough the item is part of a validated instrument[47] future studies with a complete spirituality scalecould be used to further evaluate the relationbetween spirituality and missing appointments Fifthwe do not know if the caregiver may have answeredsurvey questions about themselves in response to theprompt youyour child However coordinators whoadministered the surveys did not report that caregiversexpressed confusion with the survey instructions in this

regard Finally we were limited in what variables wecould evaluate in the present study because we wereusing an existing survey Insurance coverage anotherpotentially important variable in health care utilizationwas not captured in these surveys Also statisticallysome confidence intervals in the regression modelswere very wide suggesting a great deal of variabilitywithin the sample on some variables Direct evaluationof other components of the HBM and potentially otherimportant characteristics of participants was not poss-ible Further research is needed on the impact of rel-evant variables such as cognitive impairment andaccess to quality care on missed appointments forpopulations of individuals with SCD and their families

Conclusions

Our results demonstrated that modifiers of the HBMinfluence missed clinic appointments among individ-uals with SCD across the US Our findings supportthe importance of understanding social determinantsof health and other variables from the HBM inkeeping clinic appointments and highlight strategiesindividuals with SCD believed would help them withkeeping clinic appointments Among strategies remin-ders that are personalized to voice or text messagesbased on preferences improving the ability to get to

Table 4 Logistic regression model for missed appointment with variables from the health belief modelCombined model (N = 542)

Variable Odds ratio 95 CI Pr(gt|z|)

(Intercept) 769 (265223) lt0001Age group Children (lt18 yo)a 022 (013039) lt0001Sex Female 084 (052135) 0467Education Some college or more 063 (038104) 0072dagger

PHQ score 099 (082119) 0912Ability to pay bills Very or somewhat difficult 170 (104280) 0035Literacy High 139 (076255) 0284Spirituality Not very spiritual 183 (113294) 0013Social Support High 078 (034176) 055Adult model (N = 211)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 307 (222426) 0011Age 095 (091099) 0008Sex Female 045 (015139) 0168Education Some college or more 038 (012124) 011PHQ score 109 (076157) 064Ability to pay bills Very or somewhat difficult 499 (120207) 0027Literacy High 464 (133162) 0016Spirituality Not very spiritual 142 (046431) 054Social support High 080 (014449) 0798Model for children (N = 331)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 377 (098145) 0053Age 094 (088100) 0035Sex Female 095 (055166) 087Education Some college or more 068 (038121) 0189PHQ score 095 (076119) 063Ability to pay bills Very or somewhat difficult 144 (082253) 0199Literacy High 099 (048207) 0986Spirituality Not very spiritual 167 (095291) 0073dagger

Social support High 098 (037259) 097aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesp lt 005 daggerp lt 01

HEMATOLOGY 689

and pay for appointments and appealing to a personrsquosspirituality may improve clinic attendance Our findingssuggest potential targets to improve appointmentshow rates but also suggest areas in need of furtherstudy

Acknowledgements

The authors thank the members of the Vanderbilt-MeharryCenter of Excellence in Sickle Cell Disease for their thoughtfuland helpful comments in reviewing the manuscript BerthaDavis for regulatory matters Brittany L Myers DNP RN forsupport with patient recruitment and Natasha Dean forassistance with preparation of this manuscript

Disclosure statement

No potential conflict of interest was reported by the authors

Funding

This work was supported by PCORI [grant number CDRN1501-26498]

ORCID

Robert M Cronin httporcidorg0000-0003-1916-6521Marsha Treadwell httporcidorg0000-0003-0521-1846

References

[1] Brousseau DC Panepinto JA Nimmer M et al Thenumber of people with sickle-cell disease in theUnited States national and state estimates Am JHematol 201085(1)77ndash78

[2] Mvundura M Amendah D Kavanagh PL et al Healthcare utilization and expenditures for privately and pub-licly insured children with sickle cell disease in theUnited States Pediatr Blood Cancer 200953(4)642ndash646

[3] Steiner CA Miller JL Sickle cell disease patients in UShospitals 2004 Rockville (MD) Agency for HealthcareResearch and Quality (US) 2006

[4] Yusuf HR Atrash HK Grosse SD et al Emergency depart-ment visits made by patients with sickle cell disease adescriptive study 1999ndash2007 Am J Prev Med 201038(4 Suppl)S536ndashS541 doi101016jamepre201001001PubMed PMID 20331955 PubMed Central PMCIDPMCPMC4521762

[5] Mehta SR Afenyi-Annan A Byrns PJ et al Opportunitiesto improve outcomes in sickle cell disease Am FamPhysician 2006 Jul 1574(2)303ndash310 PubMed PMID16883928

[6] Yawn BP Buchanan GR Afenyi-Annan AN et alManagement of sickle cell disease summary of the2014 evidence-based report by expert panel membersJAMA 2014312(10)1033ndash1048 doi101001jama201410517 PubMed PMID 25203083

[7] Ware RE How I use hydroxyurea to treat young patientswith sickle cell anemia Blood 2010115(26)5300ndash5311doi101182blood-2009-04-146852 PubMed PMID20223921 PubMed Central PMCID PMCPMC2902131

[8] Thornburg CD Calatroni A Telen M et al Adherence tohydroxyurea therapy in children with sickle cell anemiaJ Pediatr 2010156(3)415ndash419 doi101016jjpeds200909044 PubMed PMID 19880135 PubMed CentralPMCID PMCPMC3901082

[9] Modi AC Crosby LE Hines J et al Feasibility of web-based technology to assess adherence to clinic appoint-ments in youth with sickle cell disease J PediatrHematol Oncol 201234(3)e93ndashe96 doi101097MPH0b013e318240d531 PubMed PMID 22278205PubMed Central PMCID PMCPMC3311776

[10] Crosby LE Modi AC Lemanek KL et al Perceived bar-riers to clinic appointments for adolescents with sicklecell disease J Pediatr Hematol Oncol 200931(8)571ndash576 doi101097MPH0b013e3181acd889 PubMedPMID 19636266 PubMed Central PMCIDPMCPMC2750821 eng

[11] Bollinger LM Nire KG Rhodes MM et al Caregiversrsquo per-spectives on barriers to transcranial Doppler screeningin children with sickle-cell disease Pediatr BloodCancer 201156(1)99ndash102 doi 101002pbc22780PubMed PMID 20842753

[12] Raphael JL Shetty PB Liu H et al A critical assessmentof transcranial Doppler screening rates in a large pedi-atric sickle cell center opportunities to improve health-care quality Pediatr Blood Cancer 200851(5)647ndash651doi101002pbc21677 PubMed PMID 18623200

[13] Jacob E Childress C Nathanson JD Barriers to care andquality of primary care services in children with sicklecell disease J Adv Nurs 201672(6)1417ndash1429 doi101111jan12756 PubMed PMID 26370255

[14] Schlenz AM Boan AD Lackland DT et al Needs assess-ment for patients with sickle cell disease in SouthCarolina 2012 Public Health Rep 2016131(1)108ndash116 doi101177003335491613100117 PubMed PMID26843676 PubMed Central PMCID PMCPMC4716478

[15] Brodsky MA Rodeghier M Sanger M et al Risk factorsfor 30-day readmission in adults with sickle-celldisease Am J Med 2017 doi101016jamjmed201612010 PubMed PMID 28065771

[16] Kheirkhah P Feng Q Travis LM et al Prevalence predic-tors and economic consequences of no-shows BMCHealth Serv Res 201516533 doi101186s12913-015-1243-z PubMed PMID 26769153 PubMed CentralPMCID PMCPMC4714455 eng

[17] Davies ML Goffman RM May JH et al Large-scale no-show patterns and distributions for clinic operationalresearch Healthcare (Basel) 20164(1) doi103390healthcare4010015 PubMed PMID 27417603 PubMedCentral PMCID PMCPMC4934549 eng

[18] Janz NK Becker MH The health belief model a decadelater Health Educ Behav 198411(1)1ndash47

[19] Stretcher VJ Rosenstock IM The health belief model InBaum A editor Cambridge handbook of psychologyhealth and medicine Cambridge CambridgeUniversity Press 1997 p 113ndash117 ISBN 0521430739

[20] Campbell B Staley D Matas M Who misses appoint-ments An empirical analysis Can J Psychiatry 199136(3)223ndash225 PubMed PMID 2059940

[21] Dove HG Schneider KC The usefulness of patientsrsquo indi-vidual characteristics in predicting no-shows in outpati-ent clinics Med Care 198119(7)734ndash740 PubMedPMID 7266121

[22] Goldman L Freidin R Cook EF et al A multivariateapproach to the prediction of no-show behavior in aprimary care center Arch Intern Med 1982142(3)563ndash567 PubMed PMID 7065791

690 R M CRONIN ET AL

HBM that were available to us were associated withmissed appointments for individuals with SCD Chil-dren and adults with SCD differed with regard to modi-fiers of the HBM that were associated with theoutcome indicating potentially different targets forimproving clinic appointment keeping Participantsalso thought that having better cues to action likereminders for clinic appointments would improvetheir attendance Our findings are consistent with find-ings from other research that have examined barriersand facilitators to appointment keeping for adoles-cents with SCD and caregivers of children with SCD[910] however research concerning appointmentkeeping for adults with SCD is limited

The CDRN data collection allowed for the first timeexamination of factors associated with appointmentkeeping for adults with SCD across the US Consistentwith other research younger transition-age adults (18

to 25 years) were most likely to miss appointments[4041] The disconnect between pediatric and adultcare is a well-known gap [42] with evidence thatpatients are unprepared for differing expectations inthe adult medical world ie making appointmentswithout the assistance of family or staff Interestinglyyounger children had more missed appointmentsthan older children which may be due to caregiversof younger children needing to get to work so theymiss more appointments as the caregiver is respon-sible to get the appointment However once theyoung adult with SCD transitions to adult care theymiss more appointments because they are more ontheir own Further it was surprising that higherhealth literacy was associated with missed appoint-ments for adults in our study Upon closer examinationcommon reasons that individuals with high health lit-eracy missed appointments were because they didnot want to miss school or work and they did notknow or forgot they had an appointment Adults withhigher literacy may have more responsibilities (egschool and work) and if they are in better health andperceive that other aspects of their lives are moreimportant they may not think they needed to beseen Further study is needed on the role that health lit-eracy plays in appointment keeping and other positivehealth behaviours for patients with SCD

We included spirituality as a modifier of the HBM inthe present study In a recent review [43] the impor-tance of spiritualityreligiosity in relation to improvedcoping and decreased health care utilization forpatients with SCD was supported We found significant

Table 1 Socio-demographics for participants with sickle cell disease (N ndash 542)

Variable Adults (N = 211)Childrena

(N = 331)Combined(N = 542)

Age Years (SD Range) 270 (180ndash700) 100 (00ndash170) 150 (00ndash700)Sex Male 91 (431)b 161 (486) 252 (465)

Female 120 (569) 170 (514) 290 (535)Raceethnicity Black African American African

or Afro-Caribbean203 (962) 324 (979) 527 (972)

Hispanic Latino or Spanish origin 5 (24) 5 (15) 10 (18)Some other race or origin 10 (47) 9 (27) 19 (35)

Highest degree or level of school completed High school graduate or less 90 (426) 134 (405) 224 (413)Some college or beyond 116 (550) 126 (381) 242 (446)

Household size Median (range) 4 (1 ndash14) 3 (1ndash8) 4 (1ndash14)Marital status Marriedliving together 54 (256) 111 (335) 165 (304)

Unmarried 147 (697) 220 (665) 377 (696)Spiritualityreligiosity Very 75 (355) 151 (456) 226 (417)

Fairly 87 (412) 102 (308) 189 (349)Slightlynot at all 39 (185) 60 (181) 99 (183)

Difficulty paying monthly bills Not verynot at all 117 (554) 176 (532) 293 (541)SomewhatVery 93 (441) 154 (465) 247 (456)

Site Midwest regionCincinnati 11 (52) 40 (121) 51 (94)Chicago 17 (81) 84 (254) 101 (186)Western regionOakland 47 (223) 0 (0) 47 (87)Mid-South regionSt Jude 6 (28) 156 (471) 162 (299)UTHSCc 47 (223) 0 (0) 47 (87)Vanderbilt 83 (393) 51 (154) 134 (247)

aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesbPercentages may not add up to 100 because of missing datacUTHSC = University of Tennessee Health Science Center

Table 2 Scores on standardized measures for the participants(n = 542) with sickle cell disease

MeasureAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Patient healthquestionnaire(PHQ-2 meanSD)

146 (155) 084 (126) 108 (141)

ENRICHD socialsupportinstrument (ESSI)(n)

Poor 47 (223) 31 (94) 78 (144)Good 164 (777) 300 (906) 464 (856)

Brief healthliteracyscreening (n)

Poor 61 (289) 75 (227) 136 (251)Good 150 (711) 256 (773) 406 (749)

aCaregivers were asked to report for their children under 18 years

HEMATOLOGY 687

associations between increased reported spiritualityand missed appointments in all individuals with SCDand further exploration of strategies to explicitly tapinto this potentially important coping mechanism isneeded

Since missed appointments are costly and contrib-ute to increased acute care utilization [1617] strategiesto improve appointment show rates could decreaseadmissions and readmissions The most commonreason for missed appointments was forgetting con-sistent with previous literature [10] The HBMrsquos cuesto action could be helpful in improving adherencewith appointments (Figure 1) Three quarters of partici-pants thought that reminders would be of help to themin making it to appointments The most common waythey wanted reminders was through phone callsHowever for adults aged 18 to 30 years comparedwith older adults text message reminders were pre-ferred (text message 63 phone call 57) demon-strating that different age groups prefer differenttypes of reminders A recent systematic review andmeta-analysis looked at reminders for clinic appoint-ments [44] It was found that people receiving notifica-tions improved clinic attendance by 23 However itwas also found that multiple notifications were signifi-cantly more effective than single notifications andvoice notifications were more effective than text

notifications Therefore clinic reminders which arenot done consistently at all the sites in this studycould help improve attendance

Among the factors that influenced missed appoint-ments in adults financial insecurity seemed to haveplayed a major role in missing appointments as adultswith SCD who had difficulty paying bills were fivetimesmore likely tomiss an appointment Financial inse-curity would be consistent with our findings that partici-pants missed appointments because they couldnrsquot getto the appointment (transportation issues) didnrsquot havethe money or did not have health insurance or co-payfor the appointment Improving the ability to get toappointments and meet expenses associated withappointments could be an important focus in improvingclinic attendance These findingsmay also highlight cog-nitive challenges [45] and vulnerability of the SCD popu-lation There is recognition that cognitive impairment inadults with SCDmay contribute to the risk of unemploy-ment while depression has been correlated to thechronic unpredictable pain events and psychosocial dis-tress associated with SCD [46] Providersmust be vigilantthat individuals with SCD and cognitive impairmentandor those with lower income and depressive symp-toms may require more attention to increase outpatientand decrease acute healthcare utilization whereappropriate

Table 3 Responses to questions about appointment keeping (n = 542)

Question ResponseAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Have youyour child missed an appointment forany reason over the past year

Yes 183 (867) 216 (653) 399 (736)No 26 (123) 114 (344) 140 (258)

Reasons youyour child missed an appointment Forgot 65 (308) 56 (259) 121 (303)Time did not work 53 (251) 42 (194) 95 (238)No way to get to the appointment 44 (209) 50 (231) 94 (236)Health impacted ability to make the appointment 40 (190) 9 (42) 49 (123)Did not know about the appointment 39 (185) 22 (102) 61 (153)Did not have the money to get to the appointment 28 (133) 17 (79) 45 (113)Had other things that needed to be done 23 (109) 12 (56) 35 (88)Couldnrsquot get off work to come to the appointment 22 (104) 34 (157) 56 (14)Family emergency 18 (85) 38 (176) 56 (14)Feeling fine and didnrsquot need to come to the appointment 11 (52) 5 (23) 16 (4)Didnrsquot want to miss workschool 10 (47) 25 (116) 35 (88)Did not have health insurance or co-pay 10 (47) 3 (14) 13 (33)Had a negative experience with previous appointment orlengthy wait times

5 (24) 0 (0) 5 (13)

Missed appointment without calling ahead No 110 (521) 247 (746) 357 (659)Yes 99 (469) 83 (251) 182 (336)

Reasons for not calling Forgot 62 (294) 44 (444) 106 (582)Did not know about the appointment 21 (100) 21 (420) 42 (231)Could not get anyone on the phone 11 (52) 10 (161) 21 (115)Did not know what number to call 5 (24) 3 (273) 8 (44)

What would help to make sure you come toeach clinic visit

A reminder 174 (825) 232 (701) 406 (749)Keeping appointments on a schedule 47 (223) 121 (366) 168 (310)Scheduling only necessary appointments instead of routineappointments

24 (114) 26 (79) 50 (92)

An incentive 12 (57) 20 (60) 32 (59)Education about need for preventative appointments 8 (38) 12 (36) 20 (37)

Preference for being reminded aboutappointments

Text message 117 (555) 170 (514) 287 (530)Phone call 131 (621) 202 (610) 333 (614)E-mail 42 (199) 97 (293) 139 (256)Postal Mail 33 (156) 120 (363) 153 (282)

Best schedule for reminder Day of or before appointment 94 (445) 128 (387) 222 (410)5 to 7 days before appointment 65 (308) 108 (326) 173 (319)All of the above 46 (218) 94 (284) 140 (258)

aCaregivers were asked to report for their children under 18 years

688 R M CRONIN ET AL

Several limitations in interpreting these results arenoted First findings are based on survey self-reportswhich can lead to recall bias Although the majorityof adults with SCD and children as reported by theircaregivers had missed an appointment informationabout missing appointments was based on self-reportonly Future studies should validate self-reports aboutmissing appointments with the electronic healthrecord Second while we were able to include respon-dents from three regions of the US in the surveyresponses may differ from other regions Third thiswas a convenience sample of participants who pre-sented to clinical sites providing SCD care While theirresponses may or may not be representative of thebroader population of patients with SCD and theirfamilies the clinical sites themselves did range fromacademic medical centres to community clinicsFourth spirituality was measured with a single-itemAlthough the item is part of a validated instrument[47] future studies with a complete spirituality scalecould be used to further evaluate the relationbetween spirituality and missing appointments Fifthwe do not know if the caregiver may have answeredsurvey questions about themselves in response to theprompt youyour child However coordinators whoadministered the surveys did not report that caregiversexpressed confusion with the survey instructions in this

regard Finally we were limited in what variables wecould evaluate in the present study because we wereusing an existing survey Insurance coverage anotherpotentially important variable in health care utilizationwas not captured in these surveys Also statisticallysome confidence intervals in the regression modelswere very wide suggesting a great deal of variabilitywithin the sample on some variables Direct evaluationof other components of the HBM and potentially otherimportant characteristics of participants was not poss-ible Further research is needed on the impact of rel-evant variables such as cognitive impairment andaccess to quality care on missed appointments forpopulations of individuals with SCD and their families

Conclusions

Our results demonstrated that modifiers of the HBMinfluence missed clinic appointments among individ-uals with SCD across the US Our findings supportthe importance of understanding social determinantsof health and other variables from the HBM inkeeping clinic appointments and highlight strategiesindividuals with SCD believed would help them withkeeping clinic appointments Among strategies remin-ders that are personalized to voice or text messagesbased on preferences improving the ability to get to

Table 4 Logistic regression model for missed appointment with variables from the health belief modelCombined model (N = 542)

Variable Odds ratio 95 CI Pr(gt|z|)

(Intercept) 769 (265223) lt0001Age group Children (lt18 yo)a 022 (013039) lt0001Sex Female 084 (052135) 0467Education Some college or more 063 (038104) 0072dagger

PHQ score 099 (082119) 0912Ability to pay bills Very or somewhat difficult 170 (104280) 0035Literacy High 139 (076255) 0284Spirituality Not very spiritual 183 (113294) 0013Social Support High 078 (034176) 055Adult model (N = 211)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 307 (222426) 0011Age 095 (091099) 0008Sex Female 045 (015139) 0168Education Some college or more 038 (012124) 011PHQ score 109 (076157) 064Ability to pay bills Very or somewhat difficult 499 (120207) 0027Literacy High 464 (133162) 0016Spirituality Not very spiritual 142 (046431) 054Social support High 080 (014449) 0798Model for children (N = 331)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 377 (098145) 0053Age 094 (088100) 0035Sex Female 095 (055166) 087Education Some college or more 068 (038121) 0189PHQ score 095 (076119) 063Ability to pay bills Very or somewhat difficult 144 (082253) 0199Literacy High 099 (048207) 0986Spirituality Not very spiritual 167 (095291) 0073dagger

Social support High 098 (037259) 097aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesp lt 005 daggerp lt 01

HEMATOLOGY 689

and pay for appointments and appealing to a personrsquosspirituality may improve clinic attendance Our findingssuggest potential targets to improve appointmentshow rates but also suggest areas in need of furtherstudy

Acknowledgements

The authors thank the members of the Vanderbilt-MeharryCenter of Excellence in Sickle Cell Disease for their thoughtfuland helpful comments in reviewing the manuscript BerthaDavis for regulatory matters Brittany L Myers DNP RN forsupport with patient recruitment and Natasha Dean forassistance with preparation of this manuscript

Disclosure statement

No potential conflict of interest was reported by the authors

Funding

This work was supported by PCORI [grant number CDRN1501-26498]

ORCID

Robert M Cronin httporcidorg0000-0003-1916-6521Marsha Treadwell httporcidorg0000-0003-0521-1846

References

[1] Brousseau DC Panepinto JA Nimmer M et al Thenumber of people with sickle-cell disease in theUnited States national and state estimates Am JHematol 201085(1)77ndash78

[2] Mvundura M Amendah D Kavanagh PL et al Healthcare utilization and expenditures for privately and pub-licly insured children with sickle cell disease in theUnited States Pediatr Blood Cancer 200953(4)642ndash646

[3] Steiner CA Miller JL Sickle cell disease patients in UShospitals 2004 Rockville (MD) Agency for HealthcareResearch and Quality (US) 2006

[4] Yusuf HR Atrash HK Grosse SD et al Emergency depart-ment visits made by patients with sickle cell disease adescriptive study 1999ndash2007 Am J Prev Med 201038(4 Suppl)S536ndashS541 doi101016jamepre201001001PubMed PMID 20331955 PubMed Central PMCIDPMCPMC4521762

[5] Mehta SR Afenyi-Annan A Byrns PJ et al Opportunitiesto improve outcomes in sickle cell disease Am FamPhysician 2006 Jul 1574(2)303ndash310 PubMed PMID16883928

[6] Yawn BP Buchanan GR Afenyi-Annan AN et alManagement of sickle cell disease summary of the2014 evidence-based report by expert panel membersJAMA 2014312(10)1033ndash1048 doi101001jama201410517 PubMed PMID 25203083

[7] Ware RE How I use hydroxyurea to treat young patientswith sickle cell anemia Blood 2010115(26)5300ndash5311doi101182blood-2009-04-146852 PubMed PMID20223921 PubMed Central PMCID PMCPMC2902131

[8] Thornburg CD Calatroni A Telen M et al Adherence tohydroxyurea therapy in children with sickle cell anemiaJ Pediatr 2010156(3)415ndash419 doi101016jjpeds200909044 PubMed PMID 19880135 PubMed CentralPMCID PMCPMC3901082

[9] Modi AC Crosby LE Hines J et al Feasibility of web-based technology to assess adherence to clinic appoint-ments in youth with sickle cell disease J PediatrHematol Oncol 201234(3)e93ndashe96 doi101097MPH0b013e318240d531 PubMed PMID 22278205PubMed Central PMCID PMCPMC3311776

[10] Crosby LE Modi AC Lemanek KL et al Perceived bar-riers to clinic appointments for adolescents with sicklecell disease J Pediatr Hematol Oncol 200931(8)571ndash576 doi101097MPH0b013e3181acd889 PubMedPMID 19636266 PubMed Central PMCIDPMCPMC2750821 eng

[11] Bollinger LM Nire KG Rhodes MM et al Caregiversrsquo per-spectives on barriers to transcranial Doppler screeningin children with sickle-cell disease Pediatr BloodCancer 201156(1)99ndash102 doi 101002pbc22780PubMed PMID 20842753

[12] Raphael JL Shetty PB Liu H et al A critical assessmentof transcranial Doppler screening rates in a large pedi-atric sickle cell center opportunities to improve health-care quality Pediatr Blood Cancer 200851(5)647ndash651doi101002pbc21677 PubMed PMID 18623200

[13] Jacob E Childress C Nathanson JD Barriers to care andquality of primary care services in children with sicklecell disease J Adv Nurs 201672(6)1417ndash1429 doi101111jan12756 PubMed PMID 26370255

[14] Schlenz AM Boan AD Lackland DT et al Needs assess-ment for patients with sickle cell disease in SouthCarolina 2012 Public Health Rep 2016131(1)108ndash116 doi101177003335491613100117 PubMed PMID26843676 PubMed Central PMCID PMCPMC4716478

[15] Brodsky MA Rodeghier M Sanger M et al Risk factorsfor 30-day readmission in adults with sickle-celldisease Am J Med 2017 doi101016jamjmed201612010 PubMed PMID 28065771

[16] Kheirkhah P Feng Q Travis LM et al Prevalence predic-tors and economic consequences of no-shows BMCHealth Serv Res 201516533 doi101186s12913-015-1243-z PubMed PMID 26769153 PubMed CentralPMCID PMCPMC4714455 eng

[17] Davies ML Goffman RM May JH et al Large-scale no-show patterns and distributions for clinic operationalresearch Healthcare (Basel) 20164(1) doi103390healthcare4010015 PubMed PMID 27417603 PubMedCentral PMCID PMCPMC4934549 eng

[18] Janz NK Becker MH The health belief model a decadelater Health Educ Behav 198411(1)1ndash47

[19] Stretcher VJ Rosenstock IM The health belief model InBaum A editor Cambridge handbook of psychologyhealth and medicine Cambridge CambridgeUniversity Press 1997 p 113ndash117 ISBN 0521430739

[20] Campbell B Staley D Matas M Who misses appoint-ments An empirical analysis Can J Psychiatry 199136(3)223ndash225 PubMed PMID 2059940

[21] Dove HG Schneider KC The usefulness of patientsrsquo indi-vidual characteristics in predicting no-shows in outpati-ent clinics Med Care 198119(7)734ndash740 PubMedPMID 7266121

[22] Goldman L Freidin R Cook EF et al A multivariateapproach to the prediction of no-show behavior in aprimary care center Arch Intern Med 1982142(3)563ndash567 PubMed PMID 7065791

690 R M CRONIN ET AL

associations between increased reported spiritualityand missed appointments in all individuals with SCDand further exploration of strategies to explicitly tapinto this potentially important coping mechanism isneeded

Since missed appointments are costly and contrib-ute to increased acute care utilization [1617] strategiesto improve appointment show rates could decreaseadmissions and readmissions The most commonreason for missed appointments was forgetting con-sistent with previous literature [10] The HBMrsquos cuesto action could be helpful in improving adherencewith appointments (Figure 1) Three quarters of partici-pants thought that reminders would be of help to themin making it to appointments The most common waythey wanted reminders was through phone callsHowever for adults aged 18 to 30 years comparedwith older adults text message reminders were pre-ferred (text message 63 phone call 57) demon-strating that different age groups prefer differenttypes of reminders A recent systematic review andmeta-analysis looked at reminders for clinic appoint-ments [44] It was found that people receiving notifica-tions improved clinic attendance by 23 However itwas also found that multiple notifications were signifi-cantly more effective than single notifications andvoice notifications were more effective than text

notifications Therefore clinic reminders which arenot done consistently at all the sites in this studycould help improve attendance

Among the factors that influenced missed appoint-ments in adults financial insecurity seemed to haveplayed a major role in missing appointments as adultswith SCD who had difficulty paying bills were fivetimesmore likely tomiss an appointment Financial inse-curity would be consistent with our findings that partici-pants missed appointments because they couldnrsquot getto the appointment (transportation issues) didnrsquot havethe money or did not have health insurance or co-payfor the appointment Improving the ability to get toappointments and meet expenses associated withappointments could be an important focus in improvingclinic attendance These findingsmay also highlight cog-nitive challenges [45] and vulnerability of the SCD popu-lation There is recognition that cognitive impairment inadults with SCDmay contribute to the risk of unemploy-ment while depression has been correlated to thechronic unpredictable pain events and psychosocial dis-tress associated with SCD [46] Providersmust be vigilantthat individuals with SCD and cognitive impairmentandor those with lower income and depressive symp-toms may require more attention to increase outpatientand decrease acute healthcare utilization whereappropriate

Table 3 Responses to questions about appointment keeping (n = 542)

Question ResponseAdults

(N = 211)Childrena

(N = 331)Combined(N = 542)

Have youyour child missed an appointment forany reason over the past year

Yes 183 (867) 216 (653) 399 (736)No 26 (123) 114 (344) 140 (258)

Reasons youyour child missed an appointment Forgot 65 (308) 56 (259) 121 (303)Time did not work 53 (251) 42 (194) 95 (238)No way to get to the appointment 44 (209) 50 (231) 94 (236)Health impacted ability to make the appointment 40 (190) 9 (42) 49 (123)Did not know about the appointment 39 (185) 22 (102) 61 (153)Did not have the money to get to the appointment 28 (133) 17 (79) 45 (113)Had other things that needed to be done 23 (109) 12 (56) 35 (88)Couldnrsquot get off work to come to the appointment 22 (104) 34 (157) 56 (14)Family emergency 18 (85) 38 (176) 56 (14)Feeling fine and didnrsquot need to come to the appointment 11 (52) 5 (23) 16 (4)Didnrsquot want to miss workschool 10 (47) 25 (116) 35 (88)Did not have health insurance or co-pay 10 (47) 3 (14) 13 (33)Had a negative experience with previous appointment orlengthy wait times

5 (24) 0 (0) 5 (13)

Missed appointment without calling ahead No 110 (521) 247 (746) 357 (659)Yes 99 (469) 83 (251) 182 (336)

Reasons for not calling Forgot 62 (294) 44 (444) 106 (582)Did not know about the appointment 21 (100) 21 (420) 42 (231)Could not get anyone on the phone 11 (52) 10 (161) 21 (115)Did not know what number to call 5 (24) 3 (273) 8 (44)

What would help to make sure you come toeach clinic visit

A reminder 174 (825) 232 (701) 406 (749)Keeping appointments on a schedule 47 (223) 121 (366) 168 (310)Scheduling only necessary appointments instead of routineappointments

24 (114) 26 (79) 50 (92)

An incentive 12 (57) 20 (60) 32 (59)Education about need for preventative appointments 8 (38) 12 (36) 20 (37)

Preference for being reminded aboutappointments

Text message 117 (555) 170 (514) 287 (530)Phone call 131 (621) 202 (610) 333 (614)E-mail 42 (199) 97 (293) 139 (256)Postal Mail 33 (156) 120 (363) 153 (282)

Best schedule for reminder Day of or before appointment 94 (445) 128 (387) 222 (410)5 to 7 days before appointment 65 (308) 108 (326) 173 (319)All of the above 46 (218) 94 (284) 140 (258)

aCaregivers were asked to report for their children under 18 years

688 R M CRONIN ET AL

Several limitations in interpreting these results arenoted First findings are based on survey self-reportswhich can lead to recall bias Although the majorityof adults with SCD and children as reported by theircaregivers had missed an appointment informationabout missing appointments was based on self-reportonly Future studies should validate self-reports aboutmissing appointments with the electronic healthrecord Second while we were able to include respon-dents from three regions of the US in the surveyresponses may differ from other regions Third thiswas a convenience sample of participants who pre-sented to clinical sites providing SCD care While theirresponses may or may not be representative of thebroader population of patients with SCD and theirfamilies the clinical sites themselves did range fromacademic medical centres to community clinicsFourth spirituality was measured with a single-itemAlthough the item is part of a validated instrument[47] future studies with a complete spirituality scalecould be used to further evaluate the relationbetween spirituality and missing appointments Fifthwe do not know if the caregiver may have answeredsurvey questions about themselves in response to theprompt youyour child However coordinators whoadministered the surveys did not report that caregiversexpressed confusion with the survey instructions in this

regard Finally we were limited in what variables wecould evaluate in the present study because we wereusing an existing survey Insurance coverage anotherpotentially important variable in health care utilizationwas not captured in these surveys Also statisticallysome confidence intervals in the regression modelswere very wide suggesting a great deal of variabilitywithin the sample on some variables Direct evaluationof other components of the HBM and potentially otherimportant characteristics of participants was not poss-ible Further research is needed on the impact of rel-evant variables such as cognitive impairment andaccess to quality care on missed appointments forpopulations of individuals with SCD and their families

Conclusions

Our results demonstrated that modifiers of the HBMinfluence missed clinic appointments among individ-uals with SCD across the US Our findings supportthe importance of understanding social determinantsof health and other variables from the HBM inkeeping clinic appointments and highlight strategiesindividuals with SCD believed would help them withkeeping clinic appointments Among strategies remin-ders that are personalized to voice or text messagesbased on preferences improving the ability to get to

Table 4 Logistic regression model for missed appointment with variables from the health belief modelCombined model (N = 542)

Variable Odds ratio 95 CI Pr(gt|z|)

(Intercept) 769 (265223) lt0001Age group Children (lt18 yo)a 022 (013039) lt0001Sex Female 084 (052135) 0467Education Some college or more 063 (038104) 0072dagger

PHQ score 099 (082119) 0912Ability to pay bills Very or somewhat difficult 170 (104280) 0035Literacy High 139 (076255) 0284Spirituality Not very spiritual 183 (113294) 0013Social Support High 078 (034176) 055Adult model (N = 211)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 307 (222426) 0011Age 095 (091099) 0008Sex Female 045 (015139) 0168Education Some college or more 038 (012124) 011PHQ score 109 (076157) 064Ability to pay bills Very or somewhat difficult 499 (120207) 0027Literacy High 464 (133162) 0016Spirituality Not very spiritual 142 (046431) 054Social support High 080 (014449) 0798Model for children (N = 331)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 377 (098145) 0053Age 094 (088100) 0035Sex Female 095 (055166) 087Education Some college or more 068 (038121) 0189PHQ score 095 (076119) 063Ability to pay bills Very or somewhat difficult 144 (082253) 0199Literacy High 099 (048207) 0986Spirituality Not very spiritual 167 (095291) 0073dagger

Social support High 098 (037259) 097aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesp lt 005 daggerp lt 01

HEMATOLOGY 689

and pay for appointments and appealing to a personrsquosspirituality may improve clinic attendance Our findingssuggest potential targets to improve appointmentshow rates but also suggest areas in need of furtherstudy

Acknowledgements

The authors thank the members of the Vanderbilt-MeharryCenter of Excellence in Sickle Cell Disease for their thoughtfuland helpful comments in reviewing the manuscript BerthaDavis for regulatory matters Brittany L Myers DNP RN forsupport with patient recruitment and Natasha Dean forassistance with preparation of this manuscript

Disclosure statement

No potential conflict of interest was reported by the authors

Funding

This work was supported by PCORI [grant number CDRN1501-26498]

ORCID

Robert M Cronin httporcidorg0000-0003-1916-6521Marsha Treadwell httporcidorg0000-0003-0521-1846

References

[1] Brousseau DC Panepinto JA Nimmer M et al Thenumber of people with sickle-cell disease in theUnited States national and state estimates Am JHematol 201085(1)77ndash78

[2] Mvundura M Amendah D Kavanagh PL et al Healthcare utilization and expenditures for privately and pub-licly insured children with sickle cell disease in theUnited States Pediatr Blood Cancer 200953(4)642ndash646

[3] Steiner CA Miller JL Sickle cell disease patients in UShospitals 2004 Rockville (MD) Agency for HealthcareResearch and Quality (US) 2006

[4] Yusuf HR Atrash HK Grosse SD et al Emergency depart-ment visits made by patients with sickle cell disease adescriptive study 1999ndash2007 Am J Prev Med 201038(4 Suppl)S536ndashS541 doi101016jamepre201001001PubMed PMID 20331955 PubMed Central PMCIDPMCPMC4521762

[5] Mehta SR Afenyi-Annan A Byrns PJ et al Opportunitiesto improve outcomes in sickle cell disease Am FamPhysician 2006 Jul 1574(2)303ndash310 PubMed PMID16883928

[6] Yawn BP Buchanan GR Afenyi-Annan AN et alManagement of sickle cell disease summary of the2014 evidence-based report by expert panel membersJAMA 2014312(10)1033ndash1048 doi101001jama201410517 PubMed PMID 25203083

[7] Ware RE How I use hydroxyurea to treat young patientswith sickle cell anemia Blood 2010115(26)5300ndash5311doi101182blood-2009-04-146852 PubMed PMID20223921 PubMed Central PMCID PMCPMC2902131

[8] Thornburg CD Calatroni A Telen M et al Adherence tohydroxyurea therapy in children with sickle cell anemiaJ Pediatr 2010156(3)415ndash419 doi101016jjpeds200909044 PubMed PMID 19880135 PubMed CentralPMCID PMCPMC3901082

[9] Modi AC Crosby LE Hines J et al Feasibility of web-based technology to assess adherence to clinic appoint-ments in youth with sickle cell disease J PediatrHematol Oncol 201234(3)e93ndashe96 doi101097MPH0b013e318240d531 PubMed PMID 22278205PubMed Central PMCID PMCPMC3311776

[10] Crosby LE Modi AC Lemanek KL et al Perceived bar-riers to clinic appointments for adolescents with sicklecell disease J Pediatr Hematol Oncol 200931(8)571ndash576 doi101097MPH0b013e3181acd889 PubMedPMID 19636266 PubMed Central PMCIDPMCPMC2750821 eng

[11] Bollinger LM Nire KG Rhodes MM et al Caregiversrsquo per-spectives on barriers to transcranial Doppler screeningin children with sickle-cell disease Pediatr BloodCancer 201156(1)99ndash102 doi 101002pbc22780PubMed PMID 20842753

[12] Raphael JL Shetty PB Liu H et al A critical assessmentof transcranial Doppler screening rates in a large pedi-atric sickle cell center opportunities to improve health-care quality Pediatr Blood Cancer 200851(5)647ndash651doi101002pbc21677 PubMed PMID 18623200

[13] Jacob E Childress C Nathanson JD Barriers to care andquality of primary care services in children with sicklecell disease J Adv Nurs 201672(6)1417ndash1429 doi101111jan12756 PubMed PMID 26370255

[14] Schlenz AM Boan AD Lackland DT et al Needs assess-ment for patients with sickle cell disease in SouthCarolina 2012 Public Health Rep 2016131(1)108ndash116 doi101177003335491613100117 PubMed PMID26843676 PubMed Central PMCID PMCPMC4716478

[15] Brodsky MA Rodeghier M Sanger M et al Risk factorsfor 30-day readmission in adults with sickle-celldisease Am J Med 2017 doi101016jamjmed201612010 PubMed PMID 28065771

[16] Kheirkhah P Feng Q Travis LM et al Prevalence predic-tors and economic consequences of no-shows BMCHealth Serv Res 201516533 doi101186s12913-015-1243-z PubMed PMID 26769153 PubMed CentralPMCID PMCPMC4714455 eng

[17] Davies ML Goffman RM May JH et al Large-scale no-show patterns and distributions for clinic operationalresearch Healthcare (Basel) 20164(1) doi103390healthcare4010015 PubMed PMID 27417603 PubMedCentral PMCID PMCPMC4934549 eng

[18] Janz NK Becker MH The health belief model a decadelater Health Educ Behav 198411(1)1ndash47

[19] Stretcher VJ Rosenstock IM The health belief model InBaum A editor Cambridge handbook of psychologyhealth and medicine Cambridge CambridgeUniversity Press 1997 p 113ndash117 ISBN 0521430739

[20] Campbell B Staley D Matas M Who misses appoint-ments An empirical analysis Can J Psychiatry 199136(3)223ndash225 PubMed PMID 2059940

[21] Dove HG Schneider KC The usefulness of patientsrsquo indi-vidual characteristics in predicting no-shows in outpati-ent clinics Med Care 198119(7)734ndash740 PubMedPMID 7266121

[22] Goldman L Freidin R Cook EF et al A multivariateapproach to the prediction of no-show behavior in aprimary care center Arch Intern Med 1982142(3)563ndash567 PubMed PMID 7065791

690 R M CRONIN ET AL

Several limitations in interpreting these results arenoted First findings are based on survey self-reportswhich can lead to recall bias Although the majorityof adults with SCD and children as reported by theircaregivers had missed an appointment informationabout missing appointments was based on self-reportonly Future studies should validate self-reports aboutmissing appointments with the electronic healthrecord Second while we were able to include respon-dents from three regions of the US in the surveyresponses may differ from other regions Third thiswas a convenience sample of participants who pre-sented to clinical sites providing SCD care While theirresponses may or may not be representative of thebroader population of patients with SCD and theirfamilies the clinical sites themselves did range fromacademic medical centres to community clinicsFourth spirituality was measured with a single-itemAlthough the item is part of a validated instrument[47] future studies with a complete spirituality scalecould be used to further evaluate the relationbetween spirituality and missing appointments Fifthwe do not know if the caregiver may have answeredsurvey questions about themselves in response to theprompt youyour child However coordinators whoadministered the surveys did not report that caregiversexpressed confusion with the survey instructions in this

regard Finally we were limited in what variables wecould evaluate in the present study because we wereusing an existing survey Insurance coverage anotherpotentially important variable in health care utilizationwas not captured in these surveys Also statisticallysome confidence intervals in the regression modelswere very wide suggesting a great deal of variabilitywithin the sample on some variables Direct evaluationof other components of the HBM and potentially otherimportant characteristics of participants was not poss-ible Further research is needed on the impact of rel-evant variables such as cognitive impairment andaccess to quality care on missed appointments forpopulations of individuals with SCD and their families

Conclusions

Our results demonstrated that modifiers of the HBMinfluence missed clinic appointments among individ-uals with SCD across the US Our findings supportthe importance of understanding social determinantsof health and other variables from the HBM inkeeping clinic appointments and highlight strategiesindividuals with SCD believed would help them withkeeping clinic appointments Among strategies remin-ders that are personalized to voice or text messagesbased on preferences improving the ability to get to

Table 4 Logistic regression model for missed appointment with variables from the health belief modelCombined model (N = 542)

Variable Odds ratio 95 CI Pr(gt|z|)

(Intercept) 769 (265223) lt0001Age group Children (lt18 yo)a 022 (013039) lt0001Sex Female 084 (052135) 0467Education Some college or more 063 (038104) 0072dagger

PHQ score 099 (082119) 0912Ability to pay bills Very or somewhat difficult 170 (104280) 0035Literacy High 139 (076255) 0284Spirituality Not very spiritual 183 (113294) 0013Social Support High 078 (034176) 055Adult model (N = 211)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 307 (222426) 0011Age 095 (091099) 0008Sex Female 045 (015139) 0168Education Some college or more 038 (012124) 011PHQ score 109 (076157) 064Ability to pay bills Very or somewhat difficult 499 (120207) 0027Literacy High 464 (133162) 0016Spirituality Not very spiritual 142 (046431) 054Social support High 080 (014449) 0798Model for children (N = 331)Variable Odds ratio 95 CI Pr(gt|z|)(Intercept) 377 (098145) 0053Age 094 (088100) 0035Sex Female 095 (055166) 087Education Some college or more 068 (038121) 0189PHQ score 095 (076119) 063Ability to pay bills Very or somewhat difficult 144 (082253) 0199Literacy High 099 (048207) 0986Spirituality Not very spiritual 167 (095291) 0073dagger

Social support High 098 (037259) 097aCaregivers reported on their education marital status and financial security and reported for their children under 18 years for the other variablesp lt 005 daggerp lt 01

HEMATOLOGY 689

and pay for appointments and appealing to a personrsquosspirituality may improve clinic attendance Our findingssuggest potential targets to improve appointmentshow rates but also suggest areas in need of furtherstudy

Acknowledgements

The authors thank the members of the Vanderbilt-MeharryCenter of Excellence in Sickle Cell Disease for their thoughtfuland helpful comments in reviewing the manuscript BerthaDavis for regulatory matters Brittany L Myers DNP RN forsupport with patient recruitment and Natasha Dean forassistance with preparation of this manuscript

Disclosure statement

No potential conflict of interest was reported by the authors

Funding

This work was supported by PCORI [grant number CDRN1501-26498]

ORCID

Robert M Cronin httporcidorg0000-0003-1916-6521Marsha Treadwell httporcidorg0000-0003-0521-1846

References

[1] Brousseau DC Panepinto JA Nimmer M et al Thenumber of people with sickle-cell disease in theUnited States national and state estimates Am JHematol 201085(1)77ndash78

[2] Mvundura M Amendah D Kavanagh PL et al Healthcare utilization and expenditures for privately and pub-licly insured children with sickle cell disease in theUnited States Pediatr Blood Cancer 200953(4)642ndash646

[3] Steiner CA Miller JL Sickle cell disease patients in UShospitals 2004 Rockville (MD) Agency for HealthcareResearch and Quality (US) 2006

[4] Yusuf HR Atrash HK Grosse SD et al Emergency depart-ment visits made by patients with sickle cell disease adescriptive study 1999ndash2007 Am J Prev Med 201038(4 Suppl)S536ndashS541 doi101016jamepre201001001PubMed PMID 20331955 PubMed Central PMCIDPMCPMC4521762

[5] Mehta SR Afenyi-Annan A Byrns PJ et al Opportunitiesto improve outcomes in sickle cell disease Am FamPhysician 2006 Jul 1574(2)303ndash310 PubMed PMID16883928

[6] Yawn BP Buchanan GR Afenyi-Annan AN et alManagement of sickle cell disease summary of the2014 evidence-based report by expert panel membersJAMA 2014312(10)1033ndash1048 doi101001jama201410517 PubMed PMID 25203083

[7] Ware RE How I use hydroxyurea to treat young patientswith sickle cell anemia Blood 2010115(26)5300ndash5311doi101182blood-2009-04-146852 PubMed PMID20223921 PubMed Central PMCID PMCPMC2902131

[8] Thornburg CD Calatroni A Telen M et al Adherence tohydroxyurea therapy in children with sickle cell anemiaJ Pediatr 2010156(3)415ndash419 doi101016jjpeds200909044 PubMed PMID 19880135 PubMed CentralPMCID PMCPMC3901082

[9] Modi AC Crosby LE Hines J et al Feasibility of web-based technology to assess adherence to clinic appoint-ments in youth with sickle cell disease J PediatrHematol Oncol 201234(3)e93ndashe96 doi101097MPH0b013e318240d531 PubMed PMID 22278205PubMed Central PMCID PMCPMC3311776

[10] Crosby LE Modi AC Lemanek KL et al Perceived bar-riers to clinic appointments for adolescents with sicklecell disease J Pediatr Hematol Oncol 200931(8)571ndash576 doi101097MPH0b013e3181acd889 PubMedPMID 19636266 PubMed Central PMCIDPMCPMC2750821 eng

[11] Bollinger LM Nire KG Rhodes MM et al Caregiversrsquo per-spectives on barriers to transcranial Doppler screeningin children with sickle-cell disease Pediatr BloodCancer 201156(1)99ndash102 doi 101002pbc22780PubMed PMID 20842753

[12] Raphael JL Shetty PB Liu H et al A critical assessmentof transcranial Doppler screening rates in a large pedi-atric sickle cell center opportunities to improve health-care quality Pediatr Blood Cancer 200851(5)647ndash651doi101002pbc21677 PubMed PMID 18623200

[13] Jacob E Childress C Nathanson JD Barriers to care andquality of primary care services in children with sicklecell disease J Adv Nurs 201672(6)1417ndash1429 doi101111jan12756 PubMed PMID 26370255

[14] Schlenz AM Boan AD Lackland DT et al Needs assess-ment for patients with sickle cell disease in SouthCarolina 2012 Public Health Rep 2016131(1)108ndash116 doi101177003335491613100117 PubMed PMID26843676 PubMed Central PMCID PMCPMC4716478

[15] Brodsky MA Rodeghier M Sanger M et al Risk factorsfor 30-day readmission in adults with sickle-celldisease Am J Med 2017 doi101016jamjmed201612010 PubMed PMID 28065771

[16] Kheirkhah P Feng Q Travis LM et al Prevalence predic-tors and economic consequences of no-shows BMCHealth Serv Res 201516533 doi101186s12913-015-1243-z PubMed PMID 26769153 PubMed CentralPMCID PMCPMC4714455 eng

[17] Davies ML Goffman RM May JH et al Large-scale no-show patterns and distributions for clinic operationalresearch Healthcare (Basel) 20164(1) doi103390healthcare4010015 PubMed PMID 27417603 PubMedCentral PMCID PMCPMC4934549 eng

[18] Janz NK Becker MH The health belief model a decadelater Health Educ Behav 198411(1)1ndash47

[19] Stretcher VJ Rosenstock IM The health belief model InBaum A editor Cambridge handbook of psychologyhealth and medicine Cambridge CambridgeUniversity Press 1997 p 113ndash117 ISBN 0521430739

[20] Campbell B Staley D Matas M Who misses appoint-ments An empirical analysis Can J Psychiatry 199136(3)223ndash225 PubMed PMID 2059940

[21] Dove HG Schneider KC The usefulness of patientsrsquo indi-vidual characteristics in predicting no-shows in outpati-ent clinics Med Care 198119(7)734ndash740 PubMedPMID 7266121

[22] Goldman L Freidin R Cook EF et al A multivariateapproach to the prediction of no-show behavior in aprimary care center Arch Intern Med 1982142(3)563ndash567 PubMed PMID 7065791

690 R M CRONIN ET AL

and pay for appointments and appealing to a personrsquosspirituality may improve clinic attendance Our findingssuggest potential targets to improve appointmentshow rates but also suggest areas in need of furtherstudy

Acknowledgements

The authors thank the members of the Vanderbilt-MeharryCenter of Excellence in Sickle Cell Disease for their thoughtfuland helpful comments in reviewing the manuscript BerthaDavis for regulatory matters Brittany L Myers DNP RN forsupport with patient recruitment and Natasha Dean forassistance with preparation of this manuscript

Disclosure statement

No potential conflict of interest was reported by the authors

Funding

This work was supported by PCORI [grant number CDRN1501-26498]

ORCID

Robert M Cronin httporcidorg0000-0003-1916-6521Marsha Treadwell httporcidorg0000-0003-0521-1846

References

[1] Brousseau DC Panepinto JA Nimmer M et al Thenumber of people with sickle-cell disease in theUnited States national and state estimates Am JHematol 201085(1)77ndash78

[2] Mvundura M Amendah D Kavanagh PL et al Healthcare utilization and expenditures for privately and pub-licly insured children with sickle cell disease in theUnited States Pediatr Blood Cancer 200953(4)642ndash646

[3] Steiner CA Miller JL Sickle cell disease patients in UShospitals 2004 Rockville (MD) Agency for HealthcareResearch and Quality (US) 2006

[4] Yusuf HR Atrash HK Grosse SD et al Emergency depart-ment visits made by patients with sickle cell disease adescriptive study 1999ndash2007 Am J Prev Med 201038(4 Suppl)S536ndashS541 doi101016jamepre201001001PubMed PMID 20331955 PubMed Central PMCIDPMCPMC4521762

[5] Mehta SR Afenyi-Annan A Byrns PJ et al Opportunitiesto improve outcomes in sickle cell disease Am FamPhysician 2006 Jul 1574(2)303ndash310 PubMed PMID16883928

[6] Yawn BP Buchanan GR Afenyi-Annan AN et alManagement of sickle cell disease summary of the2014 evidence-based report by expert panel membersJAMA 2014312(10)1033ndash1048 doi101001jama201410517 PubMed PMID 25203083

[7] Ware RE How I use hydroxyurea to treat young patientswith sickle cell anemia Blood 2010115(26)5300ndash5311doi101182blood-2009-04-146852 PubMed PMID20223921 PubMed Central PMCID PMCPMC2902131

[8] Thornburg CD Calatroni A Telen M et al Adherence tohydroxyurea therapy in children with sickle cell anemiaJ Pediatr 2010156(3)415ndash419 doi101016jjpeds200909044 PubMed PMID 19880135 PubMed CentralPMCID PMCPMC3901082

[9] Modi AC Crosby LE Hines J et al Feasibility of web-based technology to assess adherence to clinic appoint-ments in youth with sickle cell disease J PediatrHematol Oncol 201234(3)e93ndashe96 doi101097MPH0b013e318240d531 PubMed PMID 22278205PubMed Central PMCID PMCPMC3311776

[10] Crosby LE Modi AC Lemanek KL et al Perceived bar-riers to clinic appointments for adolescents with sicklecell disease J Pediatr Hematol Oncol 200931(8)571ndash576 doi101097MPH0b013e3181acd889 PubMedPMID 19636266 PubMed Central PMCIDPMCPMC2750821 eng

[11] Bollinger LM Nire KG Rhodes MM et al Caregiversrsquo per-spectives on barriers to transcranial Doppler screeningin children with sickle-cell disease Pediatr BloodCancer 201156(1)99ndash102 doi 101002pbc22780PubMed PMID 20842753

[12] Raphael JL Shetty PB Liu H et al A critical assessmentof transcranial Doppler screening rates in a large pedi-atric sickle cell center opportunities to improve health-care quality Pediatr Blood Cancer 200851(5)647ndash651doi101002pbc21677 PubMed PMID 18623200

[13] Jacob E Childress C Nathanson JD Barriers to care andquality of primary care services in children with sicklecell disease J Adv Nurs 201672(6)1417ndash1429 doi101111jan12756 PubMed PMID 26370255

[14] Schlenz AM Boan AD Lackland DT et al Needs assess-ment for patients with sickle cell disease in SouthCarolina 2012 Public Health Rep 2016131(1)108ndash116 doi101177003335491613100117 PubMed PMID26843676 PubMed Central PMCID PMCPMC4716478

[15] Brodsky MA Rodeghier M Sanger M et al Risk factorsfor 30-day readmission in adults with sickle-celldisease Am J Med 2017 doi101016jamjmed201612010 PubMed PMID 28065771

[16] Kheirkhah P Feng Q Travis LM et al Prevalence predic-tors and economic consequences of no-shows BMCHealth Serv Res 201516533 doi101186s12913-015-1243-z PubMed PMID 26769153 PubMed CentralPMCID PMCPMC4714455 eng

[17] Davies ML Goffman RM May JH et al Large-scale no-show patterns and distributions for clinic operationalresearch Healthcare (Basel) 20164(1) doi103390healthcare4010015 PubMed PMID 27417603 PubMedCentral PMCID PMCPMC4934549 eng

[18] Janz NK Becker MH The health belief model a decadelater Health Educ Behav 198411(1)1ndash47

[19] Stretcher VJ Rosenstock IM The health belief model InBaum A editor Cambridge handbook of psychologyhealth and medicine Cambridge CambridgeUniversity Press 1997 p 113ndash117 ISBN 0521430739

[20] Campbell B Staley D Matas M Who misses appoint-ments An empirical analysis Can J Psychiatry 199136(3)223ndash225 PubMed PMID 2059940

[21] Dove HG Schneider KC The usefulness of patientsrsquo indi-vidual characteristics in predicting no-shows in outpati-ent clinics Med Care 198119(7)734ndash740 PubMedPMID 7266121

[22] Goldman L Freidin R Cook EF et al A multivariateapproach to the prediction of no-show behavior in aprimary care center Arch Intern Med 1982142(3)563ndash567 PubMed PMID 7065791

690 R M CRONIN ET AL

[23] Neal RD Lawlor DA Allgar V et al Missed appointmentsin general practice retrospective data analysis from fourpractices Br J Gen Pract 2001 Oct51(471)830ndash832PubMed PMID 11677708 PubMed Central PMCIDPMCPMC1314130

[24] Mellins CA Havens JF McDonnell C et al Adherence toantiretroviral medications and medical care in HIV-infected adults diagnosed with mental and substanceabuse disorders AIDS Care 200921(2)168ndash177 doi10108009540120802001705 PubMed PMID 19229685PubMed Central PMCID PMCPMC5584780 eng

[25] Rosenbloom ST Harris P Pulley J et al The mid-southclinical data research network J Am Med InformAssoc 201421(4)627ndash632 doi101136amiajnl-2014-002745 PubMed PMID 24821742 PubMed CentralPMCID PMCPMC4078290 eng

[26] Haywood CJ Lanzkron S Bediako S et al Perceived dis-crimination patient trust and adherence to medical rec-ommendations among persons with sickle cell disease JGen Intern Med 201429(12)1657ndash1662 doi101007s11606-014-2986-7 PubMed PMID 25205621 PubMedCentral PMCID PMCPMC4242876

[27] Kroenke K Spitzer RL Williams JB The patient healthquestionnaire-2 validity of a two-item depressionscreener Med Care 200341(11)1284ndash1292

[28] Vaglio JJ Conard M Poston WS et al Testing the per-formance of the ENRICHD social support instrument incardiac patients Health Qual Life Outcomes 2004224doi1011861477-7525-2-24 PubMed PMID 15142277PubMed Central PMCID PMCPMC434528

[29] Berkman LF Blumenthal J Burg M et al Effects of treat-ing depression and low perceived social support onclinical events after myocardial infarction the enhancingrecovery in coronary heart disease patients (ENRICHD)randomized trial JAMA 20032893106ndash3616

[30] Soslashrensen K Van den Broucke S Fullam J et al Health lit-eracy and public health a systematic review and inte-gration of definitions and models BMC Public Health201212(1)1

[31] Chew LD Griffin JM Partin MR et al Validation ofscreening questions for limited health literacy in alarge VA outpatient population J Gen Intern Med200823(5)561ndash566

[32] Wallace LS Rogers ES Roskos SE et al Brief reportscreening items to identify patients with limitedhealth literacy skills J Gen Intern Med 200621(8)874ndash877 doi101111j1525-1497200600532x PubMedPMID 16881950 PubMed Central PMCIDPMCPMC1831582 eng

[33] Miller WR Thoresen CE Spirituality religion and healthAn emerging research field Am Psychol 200358(1)24ndash35 PubMed PMID 12674816

[34] Swanson L Crowther M Green L et al AfricanAmericans faith and health disparities Afr Am ResPerspect 200410(1)79ndash88

[35] Harris PA Taylor R Thielke R et al Research electronicdata capture (REDCap)mdasha metadata-driven method-ology and workflow process for providing translational

research informatics support J Biomed Inform200942(2)377ndash381 doi101016jjbi200808010PubMed PMID 18929686 PubMed Central PMCIDPMCPMC2700030

[36] R Core Team Team RC R A language and environmentfor statistical computing Vienna Austria R foundationfor Statistical Computing 2005

[37] Benton TD Boyd R Ifeagwu J et al Psychiatric diagnosisin adolescents with sickle cell disease a preliminaryreport Curr Psychiatry Rep 201113(2)111ndash115

[38] Hasan SP Hashmi S Alhassen M et al Depression insickle cell disease J Natl Med Assoc 200395(7)533

[39] Jenerette C Funk M Murdaugh C Sickle cell disease astigmatizing condition that may lead to depressionIssues Ment Health Nurs 200526(10)1081ndash1101doi10108001612840500280745 PubMed PMID16284000

[40] Wojciechowski EA Hurtig A Dorn L A natural historystudy of adolescents and young adults with sickle celldisease as they transfer to adult care a need for casemanagement services J Pediatr Nurs 200217(1)18ndash27 doiS0882596302311047 [pii] PubMed PMID11891491 eng

[41] Paulukonis ST Harris WT Coates TD et al Populationbased surveillance in sickle cell disease methods find-ings and implications from the California registry andsurveillance system in hemoglobinopathies project(RuSH) Pediatr Blood Cancer 201461(12)2271ndash2276doi101002pbc25208 PubMed PMID 25176145 eng

[42] Crosby LE Quinn CT Kalinyak KA A biopsychosocialmodel for the management of patients with sickle-celldisease transitioning to adult medical care Adv Ther201532(4)293ndash305 doi101007s12325-015-0197-1PubMed PMID 25832469 PubMed Central PMCIDPMCPMC4415939 eng

[43] Clayton-Jones D Haglund K The role of spirituality andreligiosity in persons living with sickle cell diseasea review of the literature J Holist Nurs 201634(4)351ndash360 doi1011770898010115619055 PubMedPMID 26620813 eng

[44] Robotham D Satkunanathan S Reynolds J et al Usingdigital notifications to improve attendance in clinic sys-tematic review and meta-analysis BMJ Open 20166(10)e012116 doi101136bmjopen-2016-012116PubMed PMID 27798006 PubMed Central PMCIDPMCPMC5093388 eng

[45] Sanger M Jordan L Pruthi S et al Cognitive deficits areassociated with unemployment in adults with sickle cellanemia J Clin Exp Neuropsychol 201638(6)661ndash671doi1010801380339520161149153 PubMed PMID27167865 eng

[46] Edwards CL Green M Wellington CC et al Depressionsuicidal ideation and attempts in black patients withsickle cell disease J Natl Med Assoc 2009101(11)1090ndash1095 PubMed PMID 19998636 eng

[47] Kass JD Friedman R Leserman J et al Health outcomesand a new index of spiritual experience J Sci StudyRelig 199130 203ndash211

HEMATOLOGY 691