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AJSLP

Research Article

aUniversity ofbUniversity ofcCalifornia St

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Early Language Intervention UsingDistance Video-Teleconferencing: A Pilot

Study of Young Boys With FragileX Syndrome and Their Mothers

Andrea McDuffie,a Ashley Oakes,a Wendy Machalicek,b Monica Ma,a,c

Lauren Bullard,a Sarah Nelson,a and Leonard Abbedutoa

Purpose: This study examined the effects of a naturalisticparent-implemented language intervention on the use ofverbally responsive language by mothers of 6 young boyswith fragile X syndrome. The intervention included parenteducation sessions and clinician coaching delivered onsiteand by distance video-teleconferencing.Method: A single-case multiple baseline across participantswas used to examine intervention effects on maternal useof language support strategies. A nonparametric analysiswas used to evaluate the relative effectiveness of onsitecompared with distance coaching sessions.Results: Mothers increased their use of utterances thatfollowed into their child’s focus of attention and promptedchild communication acts. Intervention effects were notobserved for maternal contingent responses to childcommunication, possibly due to the limited number of

California–DavisOregon–Eugeneate University, Sacramento

ce to Andrea McDuffie: [email protected]

Wilkinsontor: Nancy Brady

ember 11, 2014ived May 11, 2015ust 19, 2015/2015_AJSLP-14-0137

n Journal of Speech-Language Pathology • Vol. 25 • 46–66 • February 2

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spontaneous communication acts children produced.Children showed moderate increases in the use of promptedcommunication acts, whereas intervention effects onspontaneous communication acts were more modest andvariable. Comparable increases in maternal strategy usewere observed during onsite and distance sessions.Conclusions: No previous study has examined a distance-delivered parent-implemented language intervention foryoung boys with fragile X syndrome. Mothers were ableto increase their use of verbally responsive language.Intervention efficacy might be enhanced by incorporatingan augmentative and alternative communication device forsome children and a more concerted focus on increasingthe frequency of child communication acts. Findingsprovide preliminary support for the efficacy of the distancedelivery format.

Responsive verbal language input from parents hasbeen found to make an important contribution toearly language development for typically develop-

ing children (e.g., Tamis-LeMonda & Bornstein, 2002) aswell as for children at risk for or experiencing developmentaldelays (e.g., McDuffie & Yoder, 2010; Siller & Sigman,2002; Warren, Brady, Sterling, Fleming, & Marquis, 2010).Responsive verbal language follows the child’s lead, is affec-tively positive, and is provided promptly and contingentlyas the parent talks about their child’s focus of attention orresponds to their child’s nonverbal or verbal communication

acts (Bornstein, Tamis-LeMonda, & Haynes, 1999). Suchparental input facilitates early language learning by makingthe correspondence between words and their referents explicitand by minimizing the cognitive and attentional resourcesthat children must devote to language learning. Increasesin parent verbal responsiveness are frequently targetedin naturalistic language intervention programs for youngchildren with intellectual disabilities (Fey et al., 2006; Yoder& Warren, 2002) as well as those with autism spectrumdisorders (ASD; Green et al., 2010; Ingersoll & Gergans,2007; Venker, McDuffie, Ellis Weismer, & Abbeduto,2012; Yoder & Stone, 2006). There are, however, no pub-lished studies that have examined the efficacy of such inter-ventions for mothers of young boys affected by fragile Xsyndrome (FXS), the leading inherited form of intellectualdisability. This is an important population to considergiven that the behavioral characteristics of young boys

Disclosure: Leonard Abbeduto has received financial support to develop andimplement outcome measures for fragile X syndrome clinical trials from F. Hoffman-LaRoche, Ltd., Roche TCRC, Inc. and Neuren Pharmaceuticals Limited. Theremaining authors have declared that no competing interests existed at the time ofpublication.

016 • Copyright © 2016 American Speech-Language-Hearing Association

earing Association User on 02/22/2016

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with FXS and their mothers are likely to contribute to in-teractions that are less than optimal for language learning.

FXS and Associated DisordersFXS results from the mutation of a single gene

(FMR1) on the X chromosome (Brown, 2002). In the fullmutation of the FMR1 gene, a repetitive sequence of trinu-cleotides (i.e., the CGG repeats) expands to 200 or morefrom its unaffected level of 54 or fewer repeats. This expan-sion leads to methylation and transcriptional silencing of thegene, reducing or completely eliminating its protein product,FMRP (Oostra & Willemsen, 2009). FMRP is critical forexperience-dependent learning through its effects on thematuration and pruning of synapses (Bhakar, Dölen, &Bear, 2012). Individuals who have 55 to 200 CGG repeatscarry the FMR1 premutation and can have reduced levelsof FMRP as well as increased—and perhaps toxic—levelsof FMR1 messenger ribonucleic acid (Tassone & Hagerman,2003). Prevalence estimates are one in 3,500 boys and menand one in 6,000 to 8,000 girls and women for the full muta-tion (Coffee et al., 2009) and one in 468 boys and men andone in 151 girls and women for the premutation (Seltzer,Baker, et al., 2012). Boys and men with the FMR1 full mu-tation tend to be more affected than girls and women giventhe protective presence of an active healthy X chromosomein girls and women. Regardless of sex, however, individualswith the FMR1 premutation or full mutation are at higherrisk for suboptimal developmental outcomes (Jacquemont,Hagerman, Hagerman, & Leehey, 2007).

Boys and Men With FXSVirtually all boys and men with the FMR1 full muta-

tion have cognitive delays, with the vast majority havingIQs between 40 and 55 (Hessl et al., 2009). In addition tocognitive delays, the behavioral phenotype, or profile ofbehaviors that is characteristically observed (J. Harris, 2002)in boys and men with this genetic disorder, includes signifi-cant delays in language development (J. Roberts, Mirrett,Anderson, Burchinal, & Neebe, 2002), even relative towhat would be expected on the basis of nonverbal develop-mental level (Abbeduto, Brady, & Kover, 2007). The ma-jority of boys and men with FXS also display hyperactivityand attentional difficulties (Scerif, Longhi, Cole, Karmiloff-Smith, & Cornish, 2012), anxiety and withdrawal (Cordeiro,Ballinger, Hagerman, & Hessl, 2011), and repetitive (Hall,Lightbody, Hirt, Rezvani, & Reiss, 2010) and challengingbehaviors, including aggression and self-injury (Leigh,Hagerman, & Hessl, 2013; Symons, Clark, Hatton, Skinner,& Bailey, 2003). Challenging behaviors emerge early in tod-dlerhood for boys with FXS (Symons et al., 2003; Symons,Byiers, Raspa, Bishop, & Bailey, 2010) and are frequentresponses to changes in routines, task demands, and thedesire for preferred items (Machalicek et al., 2014). Up to60% of boys and men with FXS display behaviors that arefrequent and severe enough to warrant a comorbid diagnosisof an ASD (Bailey et al., 1998; Demark, Feldman, &Holden, 2003; Hall et al., 2010; S. Harris et al., 2008).

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Mothers of Children With FXSAs carriers of the FMR1 premutation or full mutation

(Nolin et al., 1996), the psychological well-being of mothersof children with FXS may be affected by the mothers’ ownbiology, including an increased risk of depression andanxiety in premutation mothers, particularly in the contextof parenting stress and stressful life events (Seltzer, Barker,et al., 2012; Wheeler et al., 2014). In fact, there is evidencethat these women are more likely to exhibit symptoms ofanxiety and depression than mothers of children with otherdisabilities (Abbeduto et al., 2004; Lewis et al., 2006).Moreover, even women with the premutation who do nothave affected children have higher rates of mental healthproblems compared with the general public (Bailey, Sideris,Roberts, & Hatton, 2008; Franke et al., 1996; J. Robertset al., 2009). It should be noted, however, that the psychiatricsymptoms associated with premutation status affect onlya subset of carrier mothers. In particular, recent studieshave examined various aspects of the premutation pheno-type, including repeat length and activation ratio (i.e., theproportion of cells that produce normal levels of FMRP),and have suggested that the association between these bio-markers and psychiatric symptoms may be nonlinear; thatis, there may be increased vulnerability in girls and womenwith midrange repeats (Hartley et al., 2012; Loesch et al.,2014; J. Roberts et al., 2009). In the present context, higherlevels of maternal stress and poorer psychological well-beingare of concern because these characteristics may decreasematernal verbal responsivity and thereby increase children’srisk for language learning problems (e.g., Landry, Smith,& Swank, 2006). The delivery of a parent-mediated inter-vention may encourage more positive parent–child interac-tions and may teach mothers ways to support their children’slanguage development. The indirect effects of interventionsthat address patterns of parent–child interactions haveyet to be examined for children with FXS (Tonnsen, Cornish,Wheeler, & Roberts, 2014).

Verbal Responsivity and LanguageDevelopment in FXS

It is likely that the behavioral phenotype of boysand men with FXS contributes to their language delays inseveral ways. Repetitive behaviors, hyperarousal, andinattention may restrict interest in actively exploring theenvironment and in maintaining productive engagementwith objects (McDuffie et al., 2015). Engaging in repetitivebehaviors can occupy children’s attention such that theydo not notice, explore, and learn about the functions ofcommon objects in their environments. Hyperarousal andinattention may disrupt sustained object engagement andprevent children from learning about the properties of ob-jects and how objects can be used or combined. Escape-maintained challenging behaviors (Machalicek et al., 2014)also may interfere with meaningful participation in dailyroutines that support language learning. Last, child challeng-ing behaviors may lead mothers to be directive rather thanverbally responsive (Brady, Warren, & Sterling, 2009).

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There is evidence from longitudinal correlationalstudies of parent–child interaction, however, that childrenwith FXS do have more positive language outcomes iftheir mothers are verbally responsive (Warren et al., 2010)and if increased levels of verbal responsivity are sustainedacross childhood (Brady, Warren, Fleming, Keller, &Sterling, 2014). These and other descriptive studies focusingon other groups of children with intellectual and develop-mental disabilities (e.g., ASD; Haebig, McDuffie, & EllisWeismer, 2013; McDuffie & Yoder, 2010; Siller & Sigman,2002) provide a rationale for providing an intervention thattargets increases in maternal verbal responsiveness as a meansof supporting language development in young children.In such an intervention, parents would mediate the effectof the intervention on child communication and languageoutcomes.

Parent-Implemented Language InterventionsRelative to clinician-implemented intervention ap-

proaches, there are several advantages to training parentsso that they can, in effect, serve as the interventionist fortheir child. Parents can embed language teaching into nat-urally occurring routines and activities, thereby providingnumerous opportunities for turn taking and communicativeinteractions throughout the day (Kashinath, Woods, &Goldstein, 2006). Child learning occurs in naturalisticcontexts with naturally occurring reinforcers and thus ismore likely to generalize to new situations and to bemaintained over time (Kaiser, Hancock, & Hester, 1998).Parent-implemented programs provide a higher intensity ofexposure to intervention content than is possible in clinician-implemented treatments (Ingersoll & Gergans, 2007).Moreover, numerous studies have demonstrated that mater-nal verbal responsiveness can be increased within the contextof an intervention that includes a parent education compo-nent (e.g., Bottema-Beutel, Yoder, Hochman, & Watson,2014; Green et al., 2010; Kasari, Gulsrud, Wong, Kwon, &Locke, 2010; Kashinath et al., 2006; Yoder & Stone 2006),provided that parents receive coaching as part of theirparticipation in the intervention program (Kaiser, Hancock,& Trent, 2007).

Although very few published studies have describedthe implementation of such an intervention approach foryoung boys with FXS, Brady et al. (2009) reported on a pi-lot study in which More Than Words, a naturalistic, parent-mediated language intervention program developed by theHanen Center (Sussman, 1999), was individually deliveredto mothers of four young boys with FXS. Three of the fourmothers increased the proportion of responsive interactionsand decreased their proportion of directive interactionswith their children. Likewise, children of these three mothersshowed concomitant increases in spoken vocabulary andrates of communicative initiations. These results, in addi-tion to the results of other studies of parent-implementedinterventions, suggest that this may be a viable interventionapproach for young children with FXS.

48 American Journal of Speech-Language Pathology • Vol. 25 • 46–66


Telepractice: Intervention Delivery at a DistanceThe American Speech-Language-Hearing Association

defines telepractice as the distance delivery of professionalservices by a speech-language pathologist (SLP), accom-plished by linking the clinician to a client or professionalusing technology-based software applications. There is stilla relative dearth of research specifically examining theefficacy of distance delivery for the provision of pediatriclanguage intervention services by SLPs. To date, most tele-practice services implemented by SLPs have involved theassessment and treatment of voice disorders, stuttering, andadult neurogenic communication disorders (Mashima &Doarn, 2008; Theodoros, 2011). In addition, there arenumerous reports on the use of telepractice for the assess-ment and treatment of individuals with ASD (Boisvert,Lang, Andrianopoulos, & Boscardin, 2010). Telepracticehas been used to deliver a variety of services to individ-uals with this diagnosis, including training teachers toimplement preference assessments and functional behaviorassessments (Barretto, Wacker, Harding, Lee, & Berg,2006; Machalicek et al., 2009) as well as training teachers(Gibson, Pennington, Stenhoff, & Hopper, 2010; Vismara,Young, Stahmer, Griffith, & Rogers, 2009) and parents toimplement behavioral interventions (McDuffie et al., 2013;Vismara, McCormick, Young, Nadhan, & Monlux, 2013;Vismara, Young, & Rogers, 2012; Wainer & Ingersoll, 2011).

Relative to face-to-face delivery of early interventionservices in the clinic or at home, one of the foremost advan-tages of Internet-based delivery approaches is the potentialto reach all individuals for whom an intervention is intended(Baggett et al., 2010). Thus, the continued development oftelepractice has been driven, in part, by its cost efficiencyand the provision of equitable access to services (Theodoros,2011). In addition, telepractice can accommodate individualscheduling needs and may provide improved fidelity ofimplementation (Baggett et al., 2010). Last, it has beensuggested that telepractice may optimize the timing, intensity,and sequencing of intervention content, leading to improvedfunctional outcomes for clients.

A recent meta-analysis of parent-implemented lan-guage interventions for children between 18 and 60 monthsof age (M. Roberts & Kaiser, 2011) reported that relativelyfew early intervention activities actually were conductedwithin family homes, although this represents the settingmost frequently identified as the natural environment forfamilies of young children (Hebbler et al., 2007). The useof distance video-teleconferencing offers the potential bene-fit of supporting parents’ use of development-enhancingroutines and activities within the home—an interventionsetting that is recommended by many professionals (e.g.,Wilcox & Woods, 2011).

At the present time, there is only one published studythat examined the preliminary effectiveness of a parent-implemented early language intervention delivered by SLPsusing distance telepractice (McDuffie et al., 2013). TheMcDuffie et al. (2013) study examined the effects of a nat-uralistic language intervention on the use of three language

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support strategies by mothers of eight preschoolers with ASD.This intervention lasted 16 weeks and consisted of fourmonthly parent education sessions held onsite at a university-based clinic. Each parent education session was paired withface-to-face coaching of a play-based parent–child interaction.Twelve weekly distance coaching sessions also were imple-mented via video-teleconferencing using a laptop computerand Skype (http://www.skype.com/en). Parents increasedtheir use of comments that described their child’s currentfocus of attention and increased their contingent verbal re-sponses to child nonverbal and verbal communication acts.Parents also increased the frequency with which they usedchoices, environmental arrangement, and time delay toprompt child communication acts, and children increasedtheir frequency of prompted communication acts. Increasesin parent strategy use were observed at equivalent levelsduring both onsite and distance coaching sessions. The cur-rent study utilized the same experimental design and inter-vention model (i.e., four parent education sessions, fourface-to-face clinician coaching sessions, and 12 distancecoaching sessions) and targeted the same parent strategies(follow-in commenting, prompting child communication,and contingent responses to child communication acts) asthe McDuffie et al. (2013) study; however, the present studyfocused on boys with the FXS full mutation and their mothers.

Research QuestionsThe goals of the current study were to (a) gather pre-

liminary data on the efficacy of a naturalistic language in-tervention for young boys with FXS designed to increasematernal verbal responsiveness and (b) evaluate the effec-tiveness of combining face-to-face parent education andcoaching with parent coaching provided at a distance usingdistance video-teleconferencing. The following researchquestions were addressed:

1. Does the intervention lead to increases in maternaluse of follow-in commenting?

2. Does the intervention lead to increases in maternaluse of indirect communication prompts and child-prompted communication?

3. Does the intervention lead to increases in childspontaneous communication acts?

4. Does the intervention lead to increases in maternaluse of contingent verbal responses to childcommunication acts?

5. Does parent use of targeted strategies duringdistance coaching sessions equal or exceed strategyuse during onsite coaching sessions?

MethodParticipants and Setting

Six boys and their biological mothers participated inthe study. Families were recruited from a Listserv of familiesaffected by FXS. Children met the following eligibility

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criteria: (a) a confirmed diagnosis of full-mutation FXS,(b) between 2 and 6 years of age, (c) no two-word spokenutterances according to maternal report, (d) English asthe primary language spoken in the home, and (d) no un-corrected sensory or motor impairments severe enoughto preclude processing and responding to verbal languageinput. One child was taking a daily dose of a prescriptionmedication for attention-deficit/hyperactivity disorder, andanother child was taking a daily dose of a prescriptionmedication for anxiety. Five dyads were from the UnitedStates, and one dyad was from Canada. These six partici-pants were part of a cohort of 12 young boys with FXSwho provided the samples for three other publications(Machalicek et al., 2014; McDuffie et al., 2015; Oakes, Ma,McDuffie, Machalicek, & Abbeduto, 2014).

A battery of standardized tests and informant reportmeasures was administered to each dyad during pre- andposttreatment visits conducted at a university-based researchcenter in the western United States. Informed consent wasobtained from all mothers, and all intervention procedureswere approved by the institutional review board. Charac-teristics of each dyad at the pretreatment visit are presentedin Table 1.

Video-Teleconferencing EquipmentEquipment loaned to each family consisted of a

13.3-in., 2.4-GHz, 250-GB hard drive/SuperDrive MacBooklaptop computer (Apple, Cupertino, CA) and an externalLogitech (Romanel-sur-Morges, Switzerland) QuickCamPro 9000 camera. The camera was connected to the laptopand positioned so that the clinician could view the area inthe home where the session took place. The clinician usedthe same model of laptop and initiated video calls using thebuilt-in iSight web camera and Skype software. Distancesessions were captured using eCamm Call Recorder software(http://www.ecamm.com). Computers were connected tobroadband Internet by Ethernet cable or wireless connec-tion. Confidentiality of data transmission was securedthrough 128-bit advanced encryption.

Design of the Intervention ProgramA nonconcurrent multiple-baseline design (Christ,

2007; Harvey, May, & Kennedy, 2004; Watson & Workman,1981) across participants was used to assess the efficacyof the intervention during onsite and distance coachingsessions. In this single-case design, the investigator prede-termines the lengths of each of several baseline phasesto be implemented at different points in time (Watson &Workman, 1981). Conducting this study using concurrentbaselines across tiers (i.e., participants) would have requiredthat at least three families travel to the research clinic duringthe same time frame, which would have been difficult toaccommodate in terms of lab staffing. As each participatingdyad entered the study, they were randomly assigned to abaseline length until all predetermined baseline lengths wereutilized. In the current study, we randomly assigned two



Table 1. Characteristics of participating dyads at preintervention.

Variable Dyad 1 Dyad 2 Dyad 3 Dyad 4 Dyad 5 Dyad 6

ChildChronological age (months) 39 35 40 27 40 43Words understooda 129 325 254 336 179 233Words produceda 1 71 0 33 2 40Comprehension age equivalentb 21 32 15 17 23 32Production age equivalentb 14 20 10 17 17 20Nonverbal developmental levelc 22 25 16 20 24 23

MaternalAge 33 38 36 35 26 27IQd 98 113 100 126 108 97

aMacArthur-Bates Communicative Development Inventory: Words and Gestures (Fenson et al., 2007). Raw scores from the 396-itemvocabulary checklist are reported. bPreschool Language Scales–Fourth Edition (Zimmerman, Steiner, & Pond, 2007). Age-equivalent scoresare reported for the Auditory Comprehension and Expressive Communication subscales. cMullen Scales of Early Learning (Mullen, 1995).Age-equivalent scores are averaged across the Visual Reception and Fine Motor subscales. dKaufmann Brief Intelligence Test–SecondEdition (Kaufman & Kaufman, 2004).

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dyads each to baseline lengths of five, eight, and 11 sessions,thus forming two groups of three participants. Each groupof three participants represented a separate multiple-baselinedesign and consisted of a participant who received five,eight, or 11 baseline sessions. Although baseline data werenot collected concurrently, changes in targeted behaviorduring treatment can be considered to be functionally re-lated to the treatment because they occurred in conjunctionwith treatment procedures that were implemented at ran-domly assigned points in time (Watson & Workman, 1981).

For research questions 1 through 4, visual analysiswas used to examine changes in each dependent variable.In addition, effect sizes were calculated using nonoverlapof all pairs (NAP; Parker & Vannest, 2009), an index sum-marizing the degree of nonoverlap between baseline-phasedata and intervention-phase data. NAP represents the per-centage of data that improve across phases (Parker, Vannest,& Davis, 2011). NAP statistically is equivalent to the areaunder the curve in Receiver Operating Characteristic analy-sis or a Mann–Whitney U test in nonparametric analysis(Parker & Vannest, 2009). In calculating NAP, the denomi-nator represents thetotal number of comparisons betweenbaseline data points and intervention data points (Nb × Ni).The numerator is computed by comparing each baseline datapoint with each intervention data point and then calculatingall overlapping pairs between baseline and intervention,with ties (e.g., baseline = 5, intervention = 5) receiving.5 point and comparisons in which the value of a baselinedata point exceeds the value of an intervention data pointreceiving 1 point. The sum of points generated from over-lapping pairs is subtracted from and then divided by theproduct term: [(Nb × Ni) − Sumoverlapping pairs]/(Nb × Ni).The value of NAP ranges from .50 to 1.00. At a value ofNAP = .50, there is a 50% chance that scores from the treat-ment phase cannot be differentiated from scores from theintervention phase. According to Parker and Vannest (2009),NAP values under .65 represent weak intervention effects,values between .66 and .92 represent moderate effects, andvalues between .93 and 1.00 represent strong effects.



Nonparametric statistics were used to address researchquestion 5. In general, changes in maternal behavior wereconsidered to represent proximal changes attributable tothe intervention, whereas changes in child behavior wereconsidered to represent more distal changes. Standardizedtesting was not repeated after the conclusion of the inter-vention. Given the developmental level of the children,it would not be likely that they would make substantialchanges in standardized assessments over a period of only16 weeks.

Structure of the Intervention ProgramBaseline Sessions

Dyads 1, 2, and 3 were compared with one anotherin one multiple-baseline design, whereas Dyads 4, 5, and 6were compared with one another in an additional multiple-baseline design. Dyads 1 and 4 were the first to enter base-line, with an additional pair of dyads entering the studyeach week. Each baseline session lasted 10 min. Baselinesessions for Dyads 1 and 4 were collected during a singleday. Baseline sessions for Dyads 2, 3, 5, and 6 were collectedover two sequential days. For each baseline session, thedyad was provided with three developmentally appropriatetoys, and the mother was instructed to play with her childas she usually would. A new toy set was provided for eachbaseline session, and breaks were taken to complete othertesting or as needed. On average, breaks between baselinesessions were 30 min. Participation in the intervention wasuniformly high; mothers completed all onsite visits (explainedbelow), and only one distance session was not completedacross all six dyads.

Parent Education LessonsThe intervention provided an onsite parent education

lesson during each visit to the clinic for a total of four parenteducation lessons. Each parent education lesson lastedapproximately 90 min and was implemented by a licensedSLP (the first author) and a masters-level speech-language

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clinician (the second author) who was completing her clinicalfellowship year. The clinician also served as the coachfor each family. Materials included written handouts andaccompanying PowerPoint (Microsoft, Redmond, WA)presentations with embedded video clips. During parenteducation lessons, parents also watched and discussed videoclips of parent–child interaction from previous coachingsessions.

Lesson 1 (Intervention Week 1) introduced the useof verbal descriptions of the child’s focus of attention (i.e.,follow-in commenting), use of preferred activities, andnoncontingent reinforcement to increase engagement inplay routines. For example, if the child were putting blocksinto a container, the mother would be encouraged to saysomething such as “Red block in!” Lesson 2 (InterventionWeek 5) introduced the use of indirect strategies to promptchild communication acts (i.e., environmental arrangement,time delay, and choice making). For example, when helpingher child with a puzzle, the mother might be encouragedto keep all of the pieces in her lap and offer the child onepiece at a time while keeping that piece in sight but outof reach of the child. Lesson 3 (Intervention Week 9) intro-duced the use of contingent verbal responding to childcommunication acts (i.e., interpreting nonverbal communi-cation acts and expanding verbal communication acts).For example, if the child reached for the horse puzzle piece,the mother might be encouraged to say “Horse! Neigh!”and then give the piece to the child. Lesson 4 (InterventionWeek 13) introduced the use of interactive book readingand open-ended questions to prompt child communicationto which the parent could contingently respond by inter-preting or expanding. For example, if a specific word wasin a child’s repertoire, the parent could ask a wh– questionto elicit that word, which would then provide the parentwith an opportunity to expand.

Parent education lessons provided opportunitiesfor the clinician to use the following coaching behaviorsadapted from Friedman, Woods, and Salisbury (2012):conversation and information sharing; direct teaching ofparent knowledge or skills using print, video, or verbalinformation; demonstration of targeted parent strategiesaccompanied by explanation; and active listening and jointproblem solving and reflection with parent.

Face-to-Face Parent–Child CoachingA face-to-face clinician coaching session was provided

immediately following each onsite parent education session(Intervention Weeks 1, 5, 9, and 13). An additional onsitecoaching session, but no parent education, was providedas a booster session at the final onsite visit (InterventionWeek 17). During onsite coaching, the clinician was in theroom with the dyad and observed the mother, demonstratedstrategy use with the child, and focused on helping themother engage her child in play and recognize opportunitiesfor using the responsive verbal interaction strategies. Theclinician provided (a) suggestions for what the mother mightdo (e.g., “Try handing him one of the blocks”), (b) verbalprompts for targeted strategy use (e.g., “Go ahead and

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interpret that point” or “This would be a good time to givehim a choice”), (c) verbal models of what the parent mightsay (e.g., “Do you want cow or bear?”), and (d) reinforce-ment in the form of descriptive praise for a verbal or physi-cal act by the parent (e.g., “Great expansion of blue!”).Thus, each onsite coaching session represented a series ofongoing opportunities for the mother to practice strategy usewith her child while receiving feedback from the clinician.

Distance Parent CoachingTwelve distance sessions were provided for each dyad,

with one distance coaching session held during each weekof the month following an onsite visit (Study Weeks 2, 3,4, 6, 7, 8, 10, 11, 12, 14, 15, and 16). Distance coachingsessions were implemented by the speech-language clinicianand provided opportunities for the caregiver to practicetargeted strategies while receiving feedback from the clini-cian as the play occurred in real time in the home.

Fidelity of Clinician CoachingThe licensed SLP who implemented the parent educa-

tion lessons and the speech-language clinician who imple-mented the onsite and distance coaching sessions had beentrained during a previous parent-mediated interventionproject (McDuffie et al., 2013). To evaluate the fidelityof coaching and performance feedback procedures, twotrained observers who were not involved in deliveringthe intervention used a six-item checklist, adapted fromFriedman et al. (2012), to code the use of coaching strate-gies during parent education lessons and onsite and distancecoaching sessions. Four randomly selected sessions (oneparent education lesson, one onsite coaching session, andtwo distance coaching sessions) were coded for each dyadusing a 30-s partial interval coding procedure. Fidelity codingwas completed for 30 min of each randomly selected par-ent education lesson and 20 min of each randomly selectedonsite and distance coaching session. Mean interobserveragreement was above 80% for each item.

Intervention TargetsFollow-In Commenting

This category of verbal responsiveness was definedas parent utterances that followed into the child’s focus ofattention and verbally described what the child was doingor playing with immediately prior to the parent response(McDuffie & Yoder, 2010). Follow-in commenting does notconvey an expectation that the child provide a behavioralor communicative response to the parent.

Indirect Communication PromptingPrompting strategies included environmental arrange-

ment (Hancock & Kaiser, 2006), time delay (Warren &Kaiser, 1986), and providing opportunities for choicemaking within and between activities (McCormick, Jolivette,& Ridgley, 2003). These prompts were considered to beindirect because they did not involve imitation prompts (e.g.,“Say _____”) or questions (e.g., “What do you want?”).



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Prompted and Spontaneous Child Communication ActsPrompted child communication acts included any

child nonverbal or verbal acts of intentional communicationthat followed within 3 s of the use of environmental arrange-ment, choice making, or time delay by the parent. Spontane-ous child communication acts included all nonverbal andverbal child acts of intentional communication that were notimitated or prompted by the mother.

Parent Verbal Responses to Child Communication ActsParents were taught to respond contingently by inter-

preting nonverbal communication acts or expanding verbalcommunication acts. When interpreting, the parent usesa noun, verb, and/or function word to linguistically encodethe presumed meaning of the child’s nonverbal communi-cation act (Yoder & Warren, 2001). When expanding, theparent adds semantic or grammatical information in re-sponse to a child’s imitative, prompted, or spontaneousverbal utterance (Yoder, Spruytenburg, Edwards, & Davies,1995).

Data Collection, Coding, and ReliabilityData on maternal strategy use and child communica-

tion acts were collected during the 10-min baseline playsamples and for 10 min at the approximate midpoint ofeach face-to-face or distance coaching session. Thus, datawere collected during five onsite coaching sessions (Inter-vention Weeks 1, 5, 9, 13, and 17) and during 12 distancecoaching sessions (Intervention Weeks 2, 3, 4, 6, 7, 8, 10,11, 12, 14, 15, and 16). During data collection, the clinicianleft the room (onsite) or muted the laptop microphone(distance) and did not interact with or provide feedback tothe mother.

For both primary and reliability coding, data werecoded from videotape using ProcoderDV (http://procoder.vueinnovations.com), a personal computer–based softwareprogram that allows accurate coding of observational datafrom digital media files (Tapp, 2003). Partial interval codingutilizing a two-step coding process was used to code follow-in commenting. On the first pass through the session video,the coder determined whether the child was engaged or notengaged during each 5-s interval. On the next pass throughthe video, the coder determined whether or not the motherhad used follow-in commenting during each interval thathad been judged as engaged. Data for this variable are re-ported as the percentage of engaged intervals during whichthe mother used follow-in commenting. The remaining de-pendent variables were coded as frequency counts. Indirectprompting represents the summed frequency with whichparents used environmental arrangement, choice making,and time delay. Prompted child communication acts repre-sent the summed frequency of nonverbal and verbal childcommunication acts that followed within 3 s of a parentprompt. Spontaneous child communication acts representthe summed frequency of nonprompted verbal and non-verbal communication acts. Parent contingent verbal re-sponses represent the summed frequency with which mothers



used either interpreting or expanding within 3 s of a childcommunication act.

Dependent variables were coded from digitized video-tapes of baseline sessions and from the 10-min portion ofeach onsite and distance session during which no coach-ing was provided. The speech-language clinician completedall primary coding, and a second trained observer indepen-dently coded 20% of baseline and treatment sessions thatwere randomly selected. For engagement, an agreementwas counted when both coders agreed that the same intervalwas judged as engaged or not engaged. For follow-incommenting, both coders had to agree that this target be-havior happened within the same 5-s interval. For the re-maining dependent variables, which were frequency counts,both observers had to independently agree that a givenbehavior occurred within a 2-s window. Thus, the recordsfor both observers were compared on a point-by-point basis,and percentage agreement was calculated as number ofagreements divided by number of agreements plus disagree-ments. This quotient was then multiplied by 100 and aver-aged across all dyads. Mean interobserver agreement was92% for engaged intervals, 90% for follow-in comment-ing, 87% for indirect communication prompts, 82% forverbal responses to child communication acts, 84% forchild-prompted communication acts, 87% for child sponta-neous communication acts, and 81% for total child com-munication acts. The data presented in Table 2 representinterobserver agreement pooled across each phase of the in-tervention (baseline and treatment) for each dyad.

ResultsIntervention Effects on Maternal VerbalResponsivity and Child Communication ActsFollow-In Commenting

Follow-in commenting was introduced during thefirst onsite intervention session (Intervention Week 1).Figures 1 and 2 display the frequency of 5-s intervals dur-ing which mothers used follow-in commenting (i.e., verballydescribed their child’s focus of attention) during baseline,intervention, and 3-month follow-up phases for Dyads 1through 3 and 4 through 6, respectively. During baseline,each mother engaged in relatively low and moderately var-ied rates of follow-in commenting (follow-in commenting:M = 15.73, range = 2–32). Mothers in Dyads 1 and 4 dem-onstrated an increasing trend in use of follow-in comment-ing immediately prior to introduction of the intervention.The use of follow-in commenting immediately increased inlevel with the introduction of this strategy during the firstonsite intervention session for the mothers in Dyads 1, 3,4, and 5. For the mother in Dyad 2, use of follow-in com-menting showed a large increase in level during the seconddistance session, whereas the mother in Dyad 6 increasedher use of follow-in commenting during the first distancesession. With the exception of the mother in Dyad 4, use offollow-in commenting temporarily declined following the im-mediacy effect and then increased and remained moderately

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Table 2. Interobserver agreement for each dependent variable.

Dyad Dependent variable

Experimental phase

Baseline Intervention

M % Range M % Range

1 Parent follow-in comments 88 83–92 84 80–90Parent communication prompts 82 78–89 88 84–92Parent interprets/expands 90 88–97 92 86–96Child-prompted communication acts 88 64–94 84 80–90Child total communication acts 87 83–90 85 80–90






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variable throughout the remainder of the intervention phase.The mother in Dyad 4 used follow-in commenting at rela-tively high levels throughout the intervention phase, withthe exception of Onsite Visit 3, when contingent respondingto child communication acts was introduced. Some overlapexists between maternal use of follow-in commenting duringthe baseline and intervention phases, but an overall meanchange in level was observed for all of the mothers (follow-in commenting: M = 33.42, range = 15–57). Results weremaintained at the 3-month follow-up assessment with theexception of Dyads 5 and 6, in which the levels of follow-incommenting were comparable to baseline performance. Interms of the size of the intervention effect across dyads, themean nonoverlap index (NAP) for follow-in commentingwas 89.75%, indicating a moderate effect of the interventionon follow-in commenting. Means, ranges, and values ofNAP for maternal follow-in commenting are individuallypresented for each dyad in Table 3.

Indirect Strategies for Prompting CommunicationIndirect prompting strategies were introduced to

the mothers during the second onsite intervention session(Intervention Week 5). Figures 3 and 4 display the frequen-cies with which (a) mothers used indirect communicationprompting strategies and (b) children responded by producing

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a nonverbal or verbal communication act during baseline,intervention, and 3-month follow-up phases for Dyads 1through 3 and 4 through 6, respectively. During baseline,maternal use of indirect prompting strategies was consistentlyvery low to nonexistent across the six mothers (indirectprompting: M = 0.10, range = 0–5), and child-promptedcommunication acts were consistently observed at low tozero levels across the six children (prompted child acts:M = 0.08, range = 0–4). Within the first several weeksfollowing the introduction of this strategy during OnsiteVisit 2 (Intervention Week 5), each mother’s use of indirectprompting increased above baseline levels. Likewise, asmall to large immediacy effect was observed in promptedcommunication acts for each of the six children. Followingthe immediacy effect, responding for both mothers andchildren increased in variability, with moderate to highlevels of variability observed across dyads for the remain-der of the intervention phase. Overlap exists between base-line and intervention phases, but an overall mean changein level was observed for the mothers (indirect prompting:M = 4.15, range = 0–13) and children (prompted childacts: M = 2.86, range = 0–12) for each of the dyads. Ma-ternal use of indirect prompting strategies maintained at3 months following the intervention for each dyad. Promptedchild communication was maintained for the children in



Figure 1. Frequency of 5-s intervals with parent follow-in commenting for Dyads 1, 2, and 3.

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Dyads 1, 4, and 5; however, prompted child communicationacts at the 3-month follow-up were observed at zero ornear-zero levels for the children in Dyads 2, 3, and 6. Interms of the size of the intervention effect across dyads, themean nonoverlap index (NAP) for maternal use of indirectprompting and child-prompted communication acts was88% and 87%, respectively, both indicating a moderateeffect of the intervention. For each dyad, means, ranges,and values of NAP for parent prompts and child-promptedcommunication acts are individually presented in Table 3.

Child Spontaneous Communication ActsIntervention effects on the spontaneous production

of nonverbal and verbal communication acts by child par-ticipants were evaluated relative to the first onsite coachingsession. Means, ranges, and NAP scores for spontaneouschild communication acts are individually presented for eachchild in Table 3. During baseline, children produced low



levels of spontaneous communication acts (child spontane-ous communication: M = 2.51, range = 0–9), with a slightimprovement during intervention sessions (M = 4.24, range =0–18). However, the child in Dyad 3 had almost no com-munication acts during baseline and continued to producevery few unprompted communication acts during the inter-vention, yielding an NAP index of .55. This value suggestsalmost complete overlap of baseline and intervention data.Two children (Dyads 1 and 6) achieved NAP scores of 64%and 67%, respectively, indicating weak intervention effects.Two children (Dyads 4 and 5) produced very few sponta-neous communication acts during baseline but did slightlyincrease their production of these acts following the intro-duction of the intervention (Intervention Week 1), result-ing in NAP scores of 70% and 79%, respectively, suggestingmoderate intervention effects. Last, the child in Dyad 2,who had the most frequent production of spontaneouscommunication acts during baseline (child spontaneous

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Figure 2. Frequency of 5-s intervals with parent follow-in commenting for Dyads 4, 5, and 6.

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communication: M = 3.88, range = 1–5) increased his pro-duction of spontaneous communication acts relative to base-line (M = 7.64, range = 0–23), with an NAP index of 72%,a moderate intervention effect.

Contingent Verbal Responses to Child Communication ActsContingent responding to child communication acts

was introduced to the mothers during the third onsiteintervention session (Intervention Week 9). For Dyads 1through 3 and 4 through 6, Figures 5 and 6, respectively,display frequencies with which children produced commu-nication acts and mothers responded contingently to theseacts by either interpreting or expanding. In these figures,the value for child communication acts is a composite met-ric representing the frequency of both spontaneous andprompted communication acts (either verbal or nonverbal).

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During baseline, maternal responses to child communicationacts varied across participants (M = 2.48, range = 0–15).With the exception of Dyads 2 and 6, contingent maternalresponses during baseline closely mirrored the varied leveland trend observed for child communication acts priorto the introduction of this parent language support strategy.The child in Dyad 3 produced almost no communicationacts, thus limiting maternal opportunities to respond con-tingently. Upon introduction of the contingent respondingstrategy during the third parent education session (Inter-vention Week 9), decreased child communication and de-creased maternal responding were observed for Dyad 1,although an immediacy effect was observed for Dyads 2and 6, followed by a decelerating trend. Responding duringthe remaining intervention sessions was moderately tohighly variable, with considerable overlap between baseline



Table 3. Mean frequency and range of parent follow-in comments and communication prompts and child-prompted and spontaneouscommunication acts (nonverbal and verbal) for each dyad.

Dyad

Dependent variable

Parent follow-Incommentinga

Parent communicationpromptsb

Child-promptedcommunication actsb

Child spontaneouscommunication actsa

M frequency Range M frequency Range M frequency Range M frequency Range

1Baseline 18.00 12–27 0.11 0–1 0.00 0 6.20 3–9Intervention 34.44 17–47 2.08 0–5 1.25 0–4 6.29 0–14NAP .96 .75 .83 .64

2Baseline 17.38 7–29 0.50 0–5 0.42 0–4 3.88 1–5Intervention 31.12 14–51 2.54 0–9 1.69 0–6 7.65 0–18NAP .85 .81 .73 .72

3Baseline 12.82 2–20 0.00 0 0.00 0 0.45 0–2Intervention 29.35 12–37 3.31 0–10 2.77 0–6 0.53 0–3NAP .95 .92 .92 .55

4Baseline 12.20 3–22 0.00 0 0.00 0 0.00 0Intervention 38.76 15–53 7.23 0–17 5.15 0–12 2.71 0–9NAP .95 .96 .92 .79



Note. NAP = nonoverlap of all pairs.a17 intervention sessions. b13 intervention sessions.

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and intervention phases. Child and maternal respondingwere high at the 3-month follow-up only for Dyads 4, 5,and 6. Overall, visual inspection and consideration of NAPvalues reflect the lack of a functional relationship betweenthe intervention and contingent maternal responses to childcommunication acts for Dyads 1 and 5 and a moderateeffect of the intervention for Dyads 2, 3, 4, and 6. Table 4displays individual means, ranges, and values of NAP foreach dyad.

Efficacy of Distance Video-Teleconferencingin Delivering Parent Coaching

Means for onsite coaching sessions were derived bysumming the total frequency for each category of parentstrategy use and dividing by five (the number of onsite ses-sions). Means for distance coaching sessions were derived bysumming the total frequency for each category of parentstrategy use and dividing by 12, with the exception of Dyad 1,who participated in 11 sessions. Nonparametric Wilcoxonsigned-ranks tests failed to reveal any significant differencesbetween settings for any of the targeted parent strategies,although strategy use during distance sessions was most con-sistent for follow-in commenting. Median scores for parentuse of follow-in commenting during onsite and distance



sessions were 30.30 and 33.96, respectively (Z = −0.734,p = .463). For parent use of communication prompts, me-dian scores for onsite and distance sessions were 2.80 and2.42, respectively (Z = −1.57, p = .116). Median scores forparent verbal responses to child communication acts duringonsite and distance sessions were 7.90 and 4.46, respectively(Z = −1.57, p = .116). These findings suggest that, on aver-age, parent use of strategies introduced during onsite visitswas maintained during distance sessions.

Following the intervention, parents were mailed a15-item survey aimed at assessing the acceptability and fea-sibility of the parent education lessons as well as face-to-face and distance coaching sessions. Parents were asked toreturn their anonymous surveys in a postage-paid envelope.For each item in the survey, a 5-point Likert scale providednumerical ratings (1 = strongly disagree, 5 = strongly agree).Higher ratings reflect greater acceptability and feasibility.All six mothers returned their surveys; the overall mean ofresponses across items and dyads was 4.80/5 (see Table 5).

DiscussionDelays in spoken language and impairments in the

use of language for social communication purposes area phenotypic characteristic of boys with FXS. In addition,

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Figure 3. Frequency of parent prompts and child-prompted communication acts for Dyads 1, 2, and 3.

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some mothers of children with FXS may be predisposed tochallenges in psychological well-being that may negativelyaffect verbal responsiveness, especially in the context ofraising a child with a disability and high rates of challengingbehavior. Thus, developing approaches to optimize patternsof parent–child interaction and support mothers as theyscaffold their child’s language development is a high prior-ity for research and practice. The intervention implementedin the current was designed to teach mothers of youngboys with FXS to use a set of responsive verbal inter-action strategies to increase the amount of appropriatelyscaffolded verbal language input that each child received.In general, the findings suggest that despite the challengesfaced by these dyads, the intervention led to meaningfuland positive changes in both maternal and child targetedbehaviors.

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All of the maternal strategies targeted in the currentstudy were selected for their utility in providing increasedlevels of salient and easily processed verbal language tochildren. In follow-in commenting, the mother was encour-aged to provide verbal language input that directly corre-sponded to the child’s ongoing focus of attention (McDuffie& Yoder, 2010). We found a moderately beneficial effectof the intervention on maternal use of follow-in commenting.In fact, all mothers showed increases over baseline, althoughat different points during the intervention. Follow-in com-menting establishes a state of symbol-infused supportedjoint engagement in which the child receives valuable con-tingent linguistic information while not being requiredto contribute actively to the maintenance of this triadicinteraction state (Adamson, Bakeman, & Deckner, 2004;Adamson, Bakeman, Deckner, & Romski, 2009). Parent



Figure 4. Frequency of parent prompts and child-prompted communication acts for Dyads 4, 5, and 6.

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use of follow-in commenting has been shown to predictgains in both comprehension and spoken language out-comes for developmentally young and minimally verbalchildren with ASD (Haebig et al., 2013). Thus, the efficacyof the present intervention in increasing maternal follow-incommenting has the potential to lead to meaningful im-provement in language outcomes for developmentallyyoung children with FXS. That two of the mothers in oursample did not maintain their use of this strategy after theend of coaching, however, suggests that some mothers mayneed more instruction or practice in this strategy or more op-tions regarding situations in which to use it.

Mothers in the current study were also taught strate-gies for indirectly prompting child communication actsthrough the use of environmental arrangement, time delay,and choice making. Such indirect prompts capitalize onchild motivation to increase requests for the desired object



or to continue a highly preferred activity or routine. Wefound that the intervention led to moderate increases inmaternal use of indirect prompts over baseline, with allmothers showing and sustaining this improvement at inter-vention follow-up. Indirect prompting is likely to be espe-cially important for children with FXS because many areinfrequent communicators (Abbeduto et al., 2007), whichmeans that they provide few opportunities for their parentsto respond with the types of contingent interpretations orexpansion that help advance language learning.

In conjunction with increased maternal prompting,all of the child participants also showed an increased fre-quency of prompted communication acts over baseline.This is an important finding because, when respondingcontingently to these acts, parents have more opportunitiesto provide the rich linguistic input from which the childrencan abstract new vocabulary and combinatorial rules. At

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Figure 5. Frequency of total child communication acts and parent contingent responses for Dyads 1, 2, and 3.

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the same time, however, we found that children did notrespond to all—or even most—indirect prompts from theirmothers, and half of the children failed to maintain theirgains at follow-up. In the intervention, mothers weretaught to use prompting as a way of continuing the child’songoing play, but continuation of play might not providesufficient reinforcement for the child with FXS. Parentsmight benefit from coaching focused on successfully con-triving transitive conditioned motivating operations thatwill temporarily increase the value of the reinforcer andthe likelihood of child communication (Carbone, 2013).Children with FXS also may require more direct physicalor echoic prompts to ensure communication followingan indirect prompt, with gradual fading of the promptsvia time delay (Carbone, Sweeney-Kerwin, Attanasio, &Kasper, 2010). In addition, children with FXS who presentwith limited functional communication may benefit frommand training with manual signs that can be reliably

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prompted. Research with children with ASD suggests im-proved speech production and comprehension as a resultof using this approach (e.g., Carbone et al., 2010; Schlosser& Wendt, 2008; van der Meer, Sutherland, O’Reilly,Lancioni, & Sigafoos, 2012). Thus, future research couldfocus on the potential benefit of a presession warm-upactivity consisting of structured mand training with high-magnitude reinforcement (e.g., food or access to highlypreferred toy) followed by the naturalistic interventionsessions.

The intervention had modest and variable effects onthe children’s spontaneous (unprompted) communicationacts. Five of the children showed increased frequency ofunprompted communication acts relative to baseline duringthe intervention. For three of these children the effectswere moderate, but for two the effects were minimal.One child showed no meaningful change in unpromptedcommunication as a result of the intervention. As indicated



Figure 6. Frequency of total child communication acts and parent contingent responses for Dyads 4, 5, and 6.

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previously, it may be necessary to provide more direct scaf-folding on very direct maternal prompts for communica-tion to achieve larger increases in child (prompted andunprompted) communication acts. Nonetheless, it is nota-ble that five of the six children were induced to be morespontaneously communicative by this relatively brief inter-vention. Increased child communication means that therewill be more opportunities for parents to engage in language“teaching” via contingent responding. Moreover, it may bethat even a rather minimal improvement in communica-tiveness can have a powerful effect on parental perceptionsof the child and on the parent’s feelings of her own parent-ing competence.

The children in the intervention who produced minimalcommunication acts (either spontaneous or prompted) mayhave benefited from an augmentative communication ap-proach. Although the use of manual signs and communicative



gestures was encouraged throughout the intervention, noneof the children had access to an aided augmentative andalternative communication device through this interventionprogram, which may have served to increase both promptedand spontaneous communication acts. Last, stimulus–stimuluspairing (Esch, Carr, & Grow, 2009; Rader et al., 2014), aprocedure in which a clinician’s targeted vocalizations areproduced in child-directed language and paired with thedelivery of highly preferred items, may have been usefulin increasing the frequency of spontaneous vocalizations orverbalizations during pretreatment sessions. Through theuse of the stimulus–stimulus pairing procedure, child vocal-izations acquire reinforcing properties due to their associationwith the preferred item and become automatic reinforcersto the child who produces them. Vocal responses acquiredin this way might then be reinforced during subsequentintervention sessions.

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Table 4. Mean frequency and range of child total communication acts (spontaneous and prompted) and parentcontingent verbal responses for each dyad.

Dyad

Dependent variable

Child totalcommunication actsa

Parent contingentverbal responsesa

M frequency Range M frequency Range

1Baseline 6.31 0–15 4.54 0–15Intervention 8.88 0–12 6.44 0–10NAP .63 .90






Note. NAP = nonoverlap of all pairs.aNine intervention sessions.

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The final targeted parent language-facilitation strat-egy was the provision of verbal language input contingentlyin response to child communication acts. Thus, motherswere taught to interpret nonverbal communication actsand to expand verbal communication acts. Four of thesix mothers did increase the frequency with which they

Table 5. Results of the parent satisfaction survey.

Overall program

Benefit of the program compared with intervention involvingRecommend to other parents of children with FXSI have learned strategies that are useful when I interact withI am able to engage my child in play more easily than prior tMy child is able to communicate more successfully than prioMy child’s attention span is longer than it was prior to the inI learned information that was relevant to my child’s commuI learned information that was relevant to my child’s behavioOverall, I am satisfied with my experiences in this program.The PowerPoint presentations were useful.Overall, I was satisfied with the parent education sessions.The coaching sessions enabled me to practice the targetedOverall, I was satisfied with the onsite coaching sessions.I thought the distance coaching sessions were helpful.It was easy to use the computer for distance coaching sess

Note. 1 = strongly disagree, 3 = neither agree nor disagree

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responded. However, the number of opportunities formothers to use this strategy was limited by the number ofspontaneous and prompted child communication acts thatwere produced. We have already mentioned that moredirect prompting of child communication may have beennecessary to support child responding. Whether this level

M Range

child only 5.00 55.00 5

my child. 4.83 4–5o the intervention. 4.83 4–5r to the intervention. 4.83 4–5tervention. 4.67 4–5nication ability. 5.00 5ral challenges. 5.00 5

4.83 4–54.33 3–54.83 4–5

strategies. 4.67 4–54.50 4–54.67 4–5

ions. 5.00 5

, 5 = strongly agree.



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of parental interpretation and expansion is sufficient toboost language learning in severely impaired children suchas those studied here remains to be determined.

Maternal use of use of follow-in commenting and in-direct prompting of communication showed more robustimprovement relative to changes in responding contin-gently to child communication acts. There are two poten-tial explanations for this finding. As mentioned previously,children whose initial levels of communication were verylow showed variable levels of prompted responding anddid not show improvements in the production of spontane-ous communication acts. Thus, some mothers simply didnot have many opportunities to respond contingently tochild communication acts relative to baseline. In addition,examination of the results for contingent responding tochild communication acts (see Figures 5 and 6) suggeststhat mothers began to increase contingent responding tochild communication acts as soon as they were taughtprompting strategies in Parent Education Session 2 (Inter-vention Week 5). Thus, it does not appear that there wasfunctional independence between parent prompting andparent contingent responses to child communication acts.It is possible that, in the process of learning follow-in com-menting and indirect strategies to prompt communication,mothers became more responsive in general and began torespond to child communication acts. It also is possiblethat when mothers learned to use indirect prompting strat-egies, they also became more aware of or able to identifycommunication acts that their children produced. At leastfour of the dyads showed an abrupt increase in maternalcontingent responding during the last four sessions prior tothe introduction of the contingent responding strategy.This time period represents the onsite and distance sessionsduring which prompting strategies were introduced andtargeted. In the current study, the decision was made to in-troduce the maternal strategies one at a time to not over-whelm the mothers. In future studies, it might be preferableto introduce all three targeted strategies at one time to avoidthe lack of independence between the dependent variablesthat arose in the current study.

The fact that maternal use of targeted interventionstrategies was comparable during both onsite and distancesessions lends preliminary support to the premise that par-ent coaching sessions can be implemented effectively bymeans of distance video-teleconferencing. We had demon-strated previously the effectiveness of distance delivery ofa parent-implemented language intervention for mothers ofchildren with ASD (McDuffie et al., 2013). Although notsignificantly different between settings, the data suggestthat parent strategy use was somewhat more consistentduring onsite relative to distance sessions. Our clinical im-pression is that the clinic setting may have been less dis-tracting for the children; that is, it may have been easier tokeep children productively engaged with both the task andthe caregiver at the clinic because the testing room wassmall and contained only a few highly interesting toys. Inaddition, it is possible that mothers were more “on point”during onsite visits than they were at home, where they



may have been preoccupied with daily obligations. Overall,however, these results do suggest that distance technologyholds great promise for the delivery of early language in-terventions and that more research is needed to refine theuse of this tool and take advantage of its potential.

Limitations of the Current StudyThe current study had several limitations. Because of

the time constraints of the onsite preintervention visits, whichlasted only 2 days, several baseline sessions were collectedon the same day for each participant. Thus, the baselinedata may not have been representative of child languageperformance over a more extended period of time. How-ever, it was our clinical impression that baseline sessionsdid accurately reflect the children’s level of communicationdevelopment. As a pilot study, the intervention includedonly six mother–child dyads and provided only 16 weeklysessions over 4 months. Although mothers did learn to usenew skills, the brief time frame of the intervention mayhave constrained the ability of the mothers to practice andconsolidate newly learned skills and precluded the likeli-hood of observing more distal effects on child communica-tion. Although the desirability of using targeted strategiesin contexts other than play was discussed frequently withmothers, we did not directly sample the use of targetedstrategies in daily routines. A further limitation was that apredetermined number of coaching sessions was providedbetween each parent education lesson. It is likely that dif-ferent mothers needed different lengths of time to becomeproficient in using targeted strategies and that a criterionlevel of performance benchmarks should have been used(e.g., M. Roberts & Kaiser, 2012) prior to the introductionof new strategies. Given the short time frame of the inter-vention, we do not present standardized test scores at post-intervention because these global measures are unlikely toreflect changes in child communication that are attribut-able to the intervention. Last, although the current studyenrolled only mothers, it is important to emphasize that anintervention that included fathers might further enrich thechild’s language learning environment and might providemothers with social support in addressing the developmen-tal needs of their children.

Future DirectionsSeveral child characteristics substantially influenced

maternal ability to implement the targeted interventionstrategies successfully. Chief among these child factorswere challenging behaviors, observed both clinically andempirically (e.g., tantrums, throwing, hitting, and self-injury;Machalicek et al., 2014), that negatively affected the amountof time children were actively engaged during coachingsessions. Opportunities for mothers to use targeted strate-gies also were limited by child inattention and limited in-terest in toys. A child’s willingness to engage with objectstheoretically can provide a conceptual platform onto whichverbally responsive caregiver language can be mapped in

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support of language development (Bottema-Beutel et al.,2014). Thus, parents will have fewer opportunities to pro-vide follow-in language to the extent that children are notinterested in, or motivated to engage with, objects. In addi-tion, it was found in a previous study that the childrenenrolled in the current study all demonstrated escape andtangibly maintained challenging behaviors (e.g., hitting,crying, elopement; Machalicek et al., 2014) and limitedobject play skills (McDuffie et al., 2015). Future iterationsof the intervention should focus on decreasing challengingbehaviors and maximizing child engagement as a compo-nent of the parent-mediated intervention.

It will be important for researchers to examine whethera greater proportion of intervention sessions could be pro-vided at a distance and to determine how best to supporteach mother as she implements the language-facilitationstrategies in the home. In the current study, families wereasked to travel substantial distances to attend a monthlyonsite visit at the research center. Although none of themothers in this study missed an onsite visit, it would clearlybe more feasible in terms of effort and cost if the majorityof the intervention could be provided at a distance ratherthan through a combination of distance and onsite sessions.Improvements in access to broadband Internet and in thequality of teleconferencing software should make an inter-vention delivered completely at a distance more attainablein the near future. Last, the number, duration, and weeklydistribution of distance sessions needed to achieve optimaloutcomes and high levels of acceptability to families remainsto be determined.

Many families with FXS have more than one af-fected child. The current study was designed to provide theintervention to each mother during interaction with onlyone of her children. An important focus of future researchwill be to refine procedures for delivering the interventionas the mother interacts with more than one child becausethis approach would be of greater utility to families of chil-dren with FXS. Future research efforts should be directedtoward establishing and evaluating methods for providingdistance coaching during family routines other than play.The importance of providing children with responsive ver-bal language input during daily routines cannot be over-stated and deserves continued attention as we develop andrefine procedures for the use of distance teleconferencing.Last, clinical trials are now underway to identify pharma-cological treatments that will remediate the core deficitsof individuals with FXS (Berry-Kravis, Knox, & Hervey,2011). An important question to consider is whether theeffects of a pharmacological intervention can be moderatedby the addition of a behavioral intervention.

AcknowledgmentsThis study was funded by a grant from the National Fragile

X Foundation to Leonard Abbeduto. We extend our deepestappreciation to the families who participated so enthusiasticallyin this study and who have continued to share updates abouttheir children’s progress.

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early language intervention using distance video ......iqs between 40 and 55 (hessl et al., 2009)....

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