Search

Accurate representation of autism spectrum disorder (ASD) in fragile X syndrome (FXS) is necessary for the field. We examined classifications of ASD using three approaches-Autism Diagnostic Observation Schedule (ADOS-2; Lord et al. 2012), Childhood Autism Rating Scale (CARS2-ST; Schopler et al. 2010), and Vineland Adaptive Behavior Scales (VABS-II; Sparrow et al. 2005)-in 45 adolescents with FXS. Maladaptive items from the VABS-II plus a maternal interview were matched with Diagnostic and Statistical Manual of Mental Disorders, 5th edition (DSM-5; American Psychiatric Association 2013) criteria for ASD. Results indicated discrepant classifications. The ADOS-2 yielded the highest rate of comorbid autism (71%); CARS2-ST and VABS-II/DSM-5 yielded lower rates (38% and 42%, respectively). A singular measure of autism symptomology is insufficient to characterize autism in FXS.

Purpose The aim of this study was to investigate language growth in individuals with fragile X syndrome (FXS) from early childhood to adolescence and the influence of maternal responsivity on language growth. Method We conducted a longitudinal analysis of language development in 55 youths (44 males, 11 females) with FXS. Data collection spanned the ages of 11–216 months. We measured expressive and receptive vocabulary with standardized tests. The number of different words and mean length of utterance were obtained from language sample analyses of mother–child interactions. We also measured maternal comments (responsivity indicator) produced during the language samples and child nonverbal IQ. Results Growth models indicated that rates of number of different words and receptive vocabulary were related to maternal commenting. Mean length of utterance did not change significantly over time. Expressive vocabulary measured with a standardized test grew, but the growth was not related to maternal commenting. Nonverbal IQ was related to all language outcomes at age of 10 years and to changes over time in vocabulary. Visual analysis indicated that the highest scores on standardized tests were produced by girls; however, measures derived from language sample analyses appeared similar for boys and girls. Language models for boys only were similar to the total sample models with lower scores at age of 10 years for some outcomes. Conclusion Results of persistent language impairments for most youths with FXS suggest the need for continued, focused interventions aimed at improved language productions in addition to a responsive environment. Supplemental Material https://doi.org/10.23641/asha.13022825

PurposeLittle is known about the language abilities of adolescents and young adults with autism spectrum disorder (ASD) despite the importance of language in their other life outcomes. Even less is known about the language abilities of racial/ethnic minorities with ASD and extensive special education needs. These gaps limit our understanding of adolescents and young adults with ASD.MethodA pilot study evaluated the efficacy of individualized age-referenced language assessment for minority adolescents and young adults with ASD in self-contained special education settings. Participants (n= 10) completed the Clinical Evaluation of Language Fundamentals–Third Edition, Test for Early Grammatical Impairment (TEGI), Columbia Mental Maturity Scale, and Wechsler Intelligence Scale for Children–Third Edition Digit Span.ResultsClinical Evaluation of Language Fundamentals–Third Edition scores showed little variation, with most participants showing a floor effect. TEGI, Columbia Mental Maturity Scale, and Digit Span scores showed greater variation. Some participants had ceiling TEGI scores, and some had variable assessment profiles.ConclusionAssessment was sensitive to variability across some measures. The pilot study outcomes support the feasibility and potential informativeness of additional investigation of conventional language assessments and change over time.

We conducted a systematic review to identify randomized controlled trials (RCTs) and single case research design (SCRD) studies of children with autism spectrum disorder that evaluate the effectiveness of responsivity intervention techniques for improving prelinguistic and/or language outcomes. Mean effect sizes were moderate and large for RCTs (33 studies; g = 0.36, 95% CI [0.21, 0.51]) and SCRD (34 studies; between-case standardized mean difference = 1.20, 95% CI [0.87, 1.54]) studies, respectively. Visual analysis (37 studies) revealed strong evidence of a functional relation for 45% of the opportunities and no evidence for 53%. Analyses of moderator effects and study quality are presented. Findings provide support for responsivity intervention strategies with more robust support for context-bound outcomes than more generalized outcomes.

In this study, we examined trajectories of specific domains of behavior problems (i.e., attention problems, depression/anxiety, and aggressive behavior) from age 6 to 18 in a sample of 55 children with fragile X syndrome. We also examined autism status and early parenting as predictors of subsequent behavioral trajectories. We found that attention problems and aggressive behavior declined steadily from childhood through adolescence whereas anxious/depressed behavior demonstrated relative stability over the same period. Youth with highly flexible mothers displayed more optional trajectories of improvement in attention problems.

Delayed and disordered speech and language development are among the primary challenges for young children with Down syndrome. Common sources of these delays include frequent ear infections and fluctuating hearing loss early in life, significant oral motor impairments, and delayed onset of joint attention and related cognitive skills. This chapter is focused on the early childhood period up to 8 years of age because most of the intervention research has been conducted during this period. The review is limited to two types of intervention: milieu teaching (MT) and augmentative and alternative communication (AAC) because much of the published research has focused on these two approaches. The review reveals the strengths and weaknesses of the research to date. Four directions for future research are proposed, most importantly combining MT and AAC approaches in a single flexible model of early communication and language intervention for young children with DS.

The predictive ability of early consonant inventory and intentional communication on later expressive language was examined in 36 boys with fragile X syndrome (FXS). Autism symptom severity was included as a potential moderator. Participants were visited in their homes twice over a 6-year period, and mother-child interactions were videotaped, coded, and transcribed behavior by behavior. Consonant inventory and concurrent autism symptom severity were predictive of later number of different words, as was the interaction between the two. Intentional communication was not predictive of number of different words. These findings provide additional specific evidence for differences in foundational language abilities associated with autism symptom severity in boys with FXS. Clinical implications are discussed.

The development of an expressive language score for people with autism based on the ADOS-2 was recently reported by Mazurek et al. (2019). The current study examined the construct validity of the ADOS-2 expressive language score (ELS) in a sample of adolescents with fragile X syndrome (n = 45, 10 girls), a neurodevelopmental disorder with high rates of autism symptomology. The ADOS-2 ELS showed strong convergent validity with multiple assessments of expressive language, receptive language, and nonverbal cognition. Divergent validity was demonstrated between the expressive language score and chronological age, symptoms of anxiety/depression, and rule-breaking behaviors. This expressive language score is a promising measure of expressive language ability that can be used in research when other language assessments are unavailable.

Background and aims The development of communicative competence requires both language and social skills. The ability to repair following a communication breakdown is critical for continued conversational interchange and to ensure comprehension of bids for communication. Communication repair demonstrates adequate language and social skills. Children with Fragile X Syndrome have difficulty with language development and social skills, which may result in delays or deficits in repair. Repair may be additionally impaired in children with Fragile X Syndrome and co-morbid autism. This study examined the development of repair in children with Fragile X Syndrome from toddlerhood into middle childhood. Methods Fifty-five children with Fragile X Syndrome and their biological mothers participated. Data were collected during in-home visits approximately every 18 months. Videotaped mother–child interactions were collected, as well as standardized assessments of language, social skills, and autism symptomology. Results Children with Fragile X Syndrome acquired the ability to repair at 90% mastery by three-and-a-half years of age. Multilevel logistic regressions predicting probability of repair indicated marginally significant effects of mean length of utterance and number of different words, and significant effects of global social skills and autism symptomology. Effect sizes were small to moderate. Conclusions Ability to repair was measured in a naturalistic setting, which allowed children with Fragile X Syndrome to utilize repairs in their daily interactions. Although children with Fragile X Syndrome may have delayed development of repair relative to typically developing expectations, in general they nonetheless catch up and demonstrate a robust ability to repair by three-and-a-half years of age. However, this study provides evidence that individual differences in language and social skills may influence ability to repair in children with Fragile X Syndrome. Finally, the relationship between autism symptoms and repair remains unclear, necessitating further exploration. Implications : Given the noted delay in repair in young children with Fragile X Syndrome, clinicians working with this population should target development of this skill as early as possible to maximize successful social interactions. This may be particularly necessary for children with Fragile X Syndrome and co-morbid autism.

Parenting children with neurodevelopmental disabilities is often challenging. Biological mothers of children with Fragile X Syndrome (FXS) may be susceptible to increased risk of mental health problems. This study examined the longitudinal relationships between maternal mental health, child challenging behaviors, and mother–child relationship quality in children and adolescents with FXS. Fifty-five mother–child dyads were followed from childhood into adolescence. The findings suggest that child challenging behaviors, maternal mental health, and mother–child relationship quality were stable during that period. Additionally, elevated levels of child challenging behaviors negatively impacted maternal mental health. Finally, child challenging behaviors, in combination with maternal mental health, influenced mother–child relationship quality. Clinical implications are discussed.

Individuals with a premutation of the fragile X mental retardation (FMR1) gene are at risk for a variety of psychological, physical, and cognitive issues, including difficulty with word retrieval. The present study examined three indicators of word retrieval difficulty; reduced productivity, reduced lexical diversity, and increased errors in word retrieval in a group of 38 female premutation carriers during standard-length speech samples collected over a period of eight years. Our results revealed that as women aged, they produced fewer words, produced fewer different words, and had greater word retrieval errors. In addition, the rate of word retrieval errors was highly correlated between two speaking contexts, indicating that this difficulty was pervasive and not solely the result of speaking in monologue. Our results suggest that subtle areas of cognitive decline emerge at a much earlier age among female premutation carriers than would be expected during healthy aging.

Purpose This research explored syntactic growth in children with fragile X syndrome (FXS) over a 5-year period, and variability in growth in relation to autism symptoms, nonverbal cognition, maternal responsivity, and gender. Method Language samples at 4 time points from 39 children with FXS, 31 boys and 8 girls, were analyzed using the Index of Productive Syntax (Scarborough, 1990) and mean length of utterance (Brown, 1973). The degree of autism symptoms was evaluated using the Childhood Autism Rating Scale (Schopler, Reichler, & Renner, 1988) at the first time point. Maternal responsivity estimates were averaged across time points. Results Children with FXS showed significant syntactic growth over time and a significant plateau (quadratic trend) in the later observations. Children who exhibited more autism symptoms at Time 1 had significantly lower syntactic abilities over time than children who exhibited fewer autism symptoms. Nonverbal cognition significantly predicted mean length of utterance scores but not Index of Productive Syntax scores. Maternal responsivity was not a significant predictor of syntactic outcomes. Girls with FXS generally demonstrated better expressive syntax than boys with FXS with notable individual differences. Conclusion Despite significant growth over time, expressive syntax is a vulnerable domain for children with FXS, especially for those with severe autism symptoms. Clinical implications arising from the current findings are discussed.

The present study examined the relationship between salivary cortisol and maternal responsiveness in mothers of boys with fragile X syndrome (FXS). Maternal responsivity is strongly associated with child outcomes, and children with FXS are at risk for compromised development due to intellectual disability and problem behavior. Increased understanding of the nature and underlying mechanisms of maternal responsivity in FXS is important to optimize outcomes in children with FXS and contribute to improved family cohesion. Data from 36 mother-child dyads indicated a complex age effect with elevated cortisol levels associated with high maternal responsivity scores when children are young and low responsivity scores when children are older. Implications of these findings and directions for future research are discussed.

OBJECTIVES: Quantity of talk and interaction in the home during early childhood is correlated with socioeconomic status (SES) and can be used to predict early language and cognitive outcomes. We tested the effectiveness of automated early language environment estimates for children 2 to 36 months old to predict cognitive and language skills 10 years later and examined effects for specific developmental age periods. METHODS: Daylong audio recordings for 146 infants and toddlers were completed monthly for 6 months, and the total number of daily adult words and adult-child conversational turnswere automatically estimated with Language Environment Analysis software. Follow-up evaluations at 9 to 14 years of age included language and cognitive testing. Language exposure for 3 age groups was assessed: 2 to 17 months, 18 to 24 months, and ≥25 months. Pearson correlations and multiple linear regression analyses were conducted. RESULTS: Conversational turn counts at 18 to 24 months of age accounted for 14% to 27% of the variance in IQ, verbal comprehension, and receptive and/or expressive vocabulary scores 10 years later after controlling for SES. Adult word counts between 18 and 24 months were correlated with language outcomes but were considerably weakened after controlling for SES. CONCLUSIONS: These data support the hypothesis that early talk and interaction, particularly during the relatively narrow developmental window of 18 to 24 months of age, can be used to predict school-age language and cognitive outcomes. With these findings, we underscore the need for effective early intervention programs that support parents in creating an optimal early language learning environment in the home.

BACKGROUND: Individuals with intellectual and developmental disabilities are more likely to engage in problem behaviours than peers with typical development. The purpose of this study was to provide descriptive and qualitative information about problem behaviours in children with fragile X syndrome (FXS) and how families respond to these behaviours. METHOD: We examined interview responses from 53 mothers of 9-year-old children with FXS. RESULTS: Defiance, tantrums, inattention, stereotypy, and aggression were the most frequently reported problem behaviours of children with FXS. Stereotypy, physical aggression, self-injury, and elopement were reported more often by mothers of children with dual diagnoses of FXS and autism than by mothers of children with a single diagnosis of FXS. CONCLUSIONS: Results indicated that for many families, by 9 years of age, problem behaviours led to frustration and altered family activities.

This study explores if children with fragile X syndrome (FXS) show advances, declines, or plateaus in adaptive behavior over time and the relationship of nonverbal cognitive abilities and autistic behavior on these trajectories. Parents of 55 children with FXS completed the Vineland Adaptive Behavior Scales (Sparrow, Balla, & Cicchetti, 1984; Sparrow, Cicchetti, & Balla, 2005) between 3 and 6 times from 2 to 10 years of age. Using raw scores, results indicate that about half of the sample showed advances in adaptive behavior, whereas the other half showed declines, indicating a regression in skills. Children who were more cognitively advanced and had less autistic behaviors had higher trajectories. Understanding the developmental course of adaptive behavior in FXS has implications for educational planning and intervention, especially for those children showing declines.

In an earlier randomized clinical trial, daily communication and language therapy resulted in more favorable spoken vocabulary outcomes than weekly therapy sessions in a subgroup of initially nonverbal preschoolers with intellectual disabilities that included only children with Down syndrome (DS). In this reanalysis of the dataset involving only the participants with DS, we found that more therapy led to larger spoken vocabularies at posttreatment because it increased children’s canonical syllabic communication and receptive vocabulary growth early in the treatment phase.

Home visiting programs support positive parenting in populations at-risk of child maltreatment, but their impact is often limited by poor retention and engagement. The current study assessed whether a cellular phone-supported version (PCI-C) of the Parent-Child Interactions (PCI) intervention improved long-term parenting practices, maternal depression, and children's aggression. Low-income mothers ( n = 371) of preschool-aged children were assigned to one of the three groups: PCI-C, PCI, and a wait-list control (WLC) group. Parenting improved in both intervention groups between baseline and 12-month follow-up compared to the WLC. Children in the PCI-C group were rated to be more cooperative and less aggressive than children in the WLC. The results offer evidence of the long-term effectiveness of PCI and the additional benefits of cellular phone supports for promoting intervention retention and improving children's behavior.

Several studies have reported declines in adaptive behavior amongst children with fragile X syndrome (FXS) starting in middle childhood. We examined the effects of maternal responsivity on adaptive behavior in 55 children with FXS visited 5-6 times in their homes from early through middle childhood. Our analyses indicated that sustained maternal responsivity had a significant positive impact on the trajectories of communication and to a lesser extent other adaptive behavior domains through middle childhood with many effects remaining significant after controlling for autism symptoms and developmental level. For children who showed declines in adaptive behavior during middle childhood, sustained high levels of maternal responsivity minimized the amount of decline observed in the communication, socialization, and daily living domains.

This study examined the efficacy of a multimodule parenting intervention, “My Baby & Me,” that began prenatally and continued until children reached 2.5 years of age. The intervention targeted specific parenting skills designed to alter trajectories of maternal and child development. Of 361 high-risk mothers (193 adolescents, 168 adults) enrolled across 4 states, half were randomly assigned to the high-intensity (HI) home visitation coaching program (55 sessions), and half to a low-intensity (LI) condition that included monthly phone calls from a coach, printed informational materials, and community resource referrals. Videotaped observations of mother–child play were coded at 5 time points for multiple maternal and child behaviors and skills. Compared to mothers in the LI group, mothers in the HI group showed higher levels of contingent responsiveness, higher quality verbal stimulation, and more verbal scaffolding by 30 months, with higher levels of warmth and greater decreases in physical intrusiveness and negativity when their children were 24 months. By 30 months, children in the HI group showed more rapid increases and higher levels of engagement with the environment, expressive language skills, and social engagement, as well as more complex toy play and fewer problem behaviors than those in the LI group. Gains in maternal responsive behaviors mediated the effects of the intervention on child outcomes. Results were comparable for adolescent and adult mothers. A strong theoretical framework, consistent focus on maternal responsiveness, high dosage, and trusting relationships with coaches are thought to explain the positive outcomes.

PurposeThis research explored whether sustained maternal responsivity (a parent–child interaction style characterized by warmth, nurturance, and stability as well as specific behaviors, such as contingent positive responses to child initiations) was a significant variable predicting vocabulary development of children with fragile X syndrome through age 9 years.MethodFifty-five mother–child dyads were followed longitudinally when children were between 2 and 10 years of age. Measures of maternal responsivity and child vocabulary were obtained at regular intervals starting at age 2.9 years. Sustained responsivity was indicated by the average responsivity measured over Observations 2–5. Responsivity at the 1st time period, autism symptoms, and cognitive development were used as control variables.ResultsAfter controlling for development and autism symptoms, the authors found significant effects for sustained responsivity on receptive vocabulary, expressive vocabulary, and the rate of different words children produced through age 9.ConclusionsMaternal responsivity, which is typically a variable of interest during early childhood, continues to be a significant variable, predicting vocabulary development through the middle childhood period. Thus, responsivity is a potential target for language interventions through this age period.

This study involves a re-analysis of spoken vocabulary outcomes of children with intellectual disabilities who were randomly assigned to receive Milieu Communication Teaching (MCT) at low (one 1-hour session per week) or high (five 1-hour sessions per week) dose frequency over nine months (Fey, Yoder, Warren, & Bredin-Oja, in press). Non-Down syndrome (NDS) and Down syndrome (DS) subgroups were matched on intelligence, mental age, and chronological age. A growth model including intercept, slope, and quadratic revealed that children in the NDS group had significantly more growth in spoken vocabulary than children in the DS group independent of dose frequency manipulations. Subsequent etiological subgroup analyses demonstrated that in the DS subgroup, children receiving MCT at the higher dose frequency had more spoken vocabulary growth than children receiving MCT at the lower dose frequency. Subgroup analyses also supported our previous findings that high dose frequency of MCT yielded greater vocabulary production outcomes than low dose frequency for children who played functionally with a range of objects, regardless of etiology.

Objective: The purpose of this study was to examine maternal responsivity and directive behaviors in mothers of children with Down syndrome (DS).Methods: Participants included 22 mothers with a young child with DS compared to 22 mothers of chronologically age-matched typically developing (TD) children using a cross-sectional design. The dyads participated in videotaped structured activities that were coded for responsive and directive behaviors.Results: Results indicated that the mothers of children with DS used a more facilitative style with the older children while these behaviors decreased with older children with TD; one directive behavior, request for behavioral comply, increased with the older children with DS.Conclusion: The mothers of children with DS adapted their parenting style to be facilitative of their children's linguistic development.

This study investigated the influence of maternal and child variables on the maternal responsivity of 55 mothers with young children with fragile X syndrome. Data included video observations of mother-child interactions in four different contexts, standardized assessments with the children, and standardized questionnaires for the mothers. The video observations were coded for child communication acts; maternal responsivity was coded at two levels: a more general measure and a behavior-by-behavior measure. Results indicated that child developmental level and language ability strongly influenced behavior-by-behavior responsivity, while maternal IQ was the strongest predictor of both general and behavior-by-behavior responsivity, after controlling for child developmental level.

Purpose The purpose of this study was to investigate the reliability of an automated language analysis system, the Language Environment Analysis (LENA), compared with a human transcriber to determine the rate of child vocalizations during recording sessions that were significantly shorter than recommended for the automated device. Method Participants were 6 nonverbal male children between the ages of 28 and 46 months. Two children had autism diagnoses, 2 had Down syndrome, 1 had a chromosomal deletion, and 1 had developmental delay. Participants were recorded by the LENA digital language processor during 14 play-based interactions with a responsive adult. Rate of child vocalizations during each of the 84 recordings was determined by both a human transcriber and the LENA software. Results A statistically significant difference between the 2 methods was observed for 4 of the 6 participants. Effect sizes were moderate to large. Variation in syllable structure did not explain the difference between the 2 methods. Vocalization rates from the 2 methods were highly correlated for 5 of the 6 participants. Conclusions Estimates of vocalization rates from nonverbal children produced by the LENA system differed from human transcription during sessions that were substantially shorter than the recommended recording length. These results confirm the recommendation of the LENA Foundation to record sessions of at least 1 hr.

Children with Down syndrome (DS) and fragile X syndrome (FXS) struggle with language development. Parenting variables, such as responsiveness to children’s communication attempts (Maternal Responsivity), and techniques used to support and teach appropriate behaviour (Behaviour Management) are known to have a significant impact on early child development. We examined these two aspects of parenting style via coded, videotaped parent-child interactions in two groups of participants matched on child age (2–5 years) and child expressive language level: mothers of children with DS and mothers of children with FXS. The mothers differed in their use of gestures and redirecting the child’s attention. Overall, mothers in both groups of children appeared to adapt appropriately to their children’s developmental needs.

Purpose The current study investigated finiteness marking (e.g., he walk s , he walk ed ) in boys with fragile X syndrome (FXS); the boys were grouped based on receptive vocabulary (i.e., borderline, impaired). Method Twenty-one boys with the full mutation of fragile X, between the ages of 8 and 16 years participated. The boys completed probes from the Test of Early Grammatical Impairment (TEGI; Rice & Wexler, 2001), a language sample, a nonverbal IQ test (Leiter–R; Roid & Miller, 1997), a receptive vocabulary test (the Pearson Picture Vocabulary Test—Fourth Edition [PPVT–IV]; Dunn & Dunn, 2007), and a measure of autistic symptoms (the Childhood Autism Rating Scale [CARS]; Schopler, Reichler, & Renner, 2002). Results There were group differences for finiteness responses on the 3rd person singular probe; the group with impaired vocabulary omitted markers with greater frequency compared to the borderline vocabulary group. There were not significant differences on the past tense probe, with both groups performing lower than expectations based on receptive vocabulary ability. Nonverbal IQ was not correlated with the measures of finiteness marking. Conclusion Boys with FXS demonstrate delays in finiteness marking, in particular, on past tense verbs. Boys with FXS show a unique profile, unlike children with SLI, in which their use of tense markers may exceed expectations benchmarked to clause length.

The purpose of this study was to examine maternal parenting styles across age-matched siblings using a within-family design, in which one child has Fragile X syndrome. Thirteen families participated; children were aged 16 to 71 months. Mothers completed several videotaped activities with each child separately as well as an interview. Mothers used a consistent, responsive style with both children, using the same degree of positive affect and warmth. Differences included using more behavior management strategies with the child with Fragile X and a conversational style of interaction with the sibling. Differences in approaches suggest the mothers adapted to the developmental differences between the children. The interview data supported these findings; mothers were aware of the changes made to accommodate the developmental differences.

The relationship between early maternal responsivity and later child communication outcomes in young children with fragile X syndrome was investigated. Data were obtained from 55 mother–child dyads over a 36-month period. Performance data were obtained at each measurement point from video observations of four different contexts. These were coded for (a) child communication behaviors, (b) parent responsivity, and (c) behavior management behaviors. Results indicate that early maternal responsivity predicts the level of four important child language outcomes at 36 months of age after controlling for child developmental level and autism symptomology.

The study compared the vocal production and language learning environments of 26 young children with autism spectrum disorder (ASD) to 78 typically developing children using measures derived from automated vocal analysis. A digital language processor and audio-processing algorithms measured the amount of adult words to children and the amount of vocalizations they produced during 12-h recording periods in their natural environments. The results indicated significant differences between typically developing children and children with ASD in the characteristics of conversations, the number of conversational turns, and in child vocalizations that correlated with parent measures of various child characteristics. Automated measurement of the language learning environment of young children with ASD reveals important differences from the environments experienced by typically developing children.

SUMMARY This study reports the effects of a neurobehavioral intervention approach on the motor skills of four children with cerebral palsy between 21 and 34 months of age. The intervention is based on the merging of neuromotor and behavioral approaches. The behavioral approach, using principles of ‘how to teach’, addressed motivational issues and allowed for precise definitions of expected outcomes which assisted in measuring acquisition and generalization of behaviors. The neuromotor approach provided ‘what to teach’ by focusing the intervention on underlying movement components (e.g., trunk rotation, weight bearing) rather than discrete milestone skills. The impact of the approach was assessed using a multiple-baseline design replicated three times. The children demonstrated the movement component by using it to perform both a treated exemplar skill (i.e. a motor skill requiring the movement component for execution) and an untreated exemplar skill. RESUME Analyse experimentale d'une approche nwtrice neurocomportementale L' ‘article rapporte les effets d'une approche neurocomportementale dans les interventions pour le developpement des capacites motrices de quartre enfants IMC âges de 21 a 34 mois. L'’ action est basee sur un mixage des approches de reeducation motrice et comportementales. L'approche comportementale. utilisant les principes de ‘comment apprendre’, repose sur la motivation et permet des definitions precises des resultats a attendre, ce qui est une aide pour apprecier les acquisitions et la generalisation des comportcments. L'approche neuromotrice fournit ‘ce qui est a apprendre’ en centrant L'intervention sur les composantes sous-jacentes de mouvement (par exemple, rotation du tronc, port de poids) plutot que sur des etapes d'acquisition predefinies. L'efficacite de l'approche a eteevaluee a partir d'un protoeole multi-criteres repete trois fois. Les enfants demontraient qu'une composante de mouvement avait bien ete acquise en y faisant appel, a la fois pour effectuer une tache apprise (par exemple une activite motrice apprise exigeant la composante de mouvement pour son execution) et dans un contexte no aborde au cours du traitement. ZUSAMMENFASSUNG Experimenielle Analyse eines verhaltensneurologischen motorischen Behanjlungskonzepies Diese Arbeit berichtet uber die Einflusse eines verhaltensneurologischen Behandlungskonzeptes auf die motorischen Fahigkeiten von vier Kindern mit Cerebralparese im Alter zwischen 21 und 34 Monaten. Die Behandlung basierte auf einer Verbindung von neuromotorischen und verhaltensneurologischen Ansatzen. Der verhaltensneurologische Ansatz, der nach den Prinzipien ‘wie wird gelchrt’ vorging, arbeitete mit der Motivation und ermoglichte genaue Definitionen des erwartelen Outcome, wodurch die Linstufung des Lrlernens und der allgemeinen Ausbreitung von Verhaltensmustern erleichtert vvurde. Der neuromoiorische Ansatz beinhaltetc ‘was wird gelehrt’, wobei sieh die Behandlung mehr auf die zugrundeliegendcn Bewegungskomponenten (z.B. Rumpfrotation, weight bearing) konzentrierte als auf diskrete als Meilensteine bekannte Fahigkeiten. Das Krgebnis des Ansatzes wurde anhand eines multiple-baseline design beurteili. Die Kinder zcigten die Bewegungskomponente, sowohl bei den behandelten exemplarischen Fahigkeiten (z.B. cine motorische Fahigkeit, die nur mit Hilfe der Bewegungskomponente ausgefuhrt werden kann), als auch bei den unbehandelten exemplarischen Fahigkeiten. RESUMEN Analisis experimental tie una intervencion minora neuroconductual Se aportan los efcctos de una intervcncion neuroconductual sobre las habilidades motoras de cuatro ninos con paralisis cerebral de 21 a 34 meses dc cdad. La intervencion se basa en la confluencia de aproximaciones neuromotoras y eonductuales. La aproximacion conduclual usando principios de ‘como ensenar’ orienta factores motivacionales y pcrmite definiciones precisas de cursos esperados que actuan en la medicion de ta adquisicion y getieralizacion de conductas. La aproximacion neuromotora aporta el ‘que ensenar’ concentrando la intervencion en determinados componentes secundarios del movimiento (por ej. rotacion del tronco, soportar un peso) mas bien que en hitos discretos. El impacto de la aproximacion se evaluo utilizando un diseno de multiples lineas basales replicada tres veces. Los ninos mostraron el componente motor utilizando lo para rcalizar una habilidad ejemplar tratada (esto es una habilidad motora que requerfa el componente motor para realizarse) y una habilidad ejemplar no tratada.

Purpose To evaluate the longitudinal effects of a 6-month course of responsivity education (RE)/prelinguistic milieu teaching (PMT) for young children with developmental delay. Method Fifty-one children, age 24–33 months, with fewer than 10 expressive words were randomly assigned to early-treatment/no-treatment groups. All treatment was added as a supplement to services that the children received in the community. Follow-up data were collected 6 and 12 months after the conclusion of the initial 6-month treatment/no-treatment conditions. Results No effects of this treatment were detected 6 or 12 months after the conclusion of the initial treatment condition. Conclusions M. E. Fey et al. (2006) reported that 6 months of RE/PMT led to a significant treatment effect in the use of intentional communication in 1 of 2 communication sampling contexts. This finding, combined with evidence from other studies, suggests that RE/PMT may be applied clinically at low intensity with the expectation of medium-sized effects on children’s rate of intentional communication acts over the short term. The results of the present study, however, provide no evidence for the anticipated longer term benefits of this intervention. Further investigation of the approach at higher intensity levels and for longer periods of time is warranted.

Objective As technology advances, newborn screening will be possible for conditions not screened today. With an expansion of screening, strategies will be needed to support family adaptation to unexpected and possibly uncertain genetic information provided shortly after birth. Method Although candidate conditions for expanded newborn screening will typically be associated with increased morbidity or mortality, for most there is no proven medical treatment that must be implemented quickly. Many will have clinical features that gradually emerge and for which the severity of impact is not predictable. Parents will seek guidance on information, support, and treatment possibilities. This article summarizes issues evoked by expanded newborn screening and suggests strategies for supporting families of identified children. Results We propose four components necessary to support family adaptation to pre-symptomatic and ‘‘untreatable’’ conditions in an era of expanded newborn screening: (1) accurate and understandable information; (2) formal and informal support; (3) active surveillance; and (4) general and targeted interventions. We argue that no condition is ‘‘untreatable’’ and that a well-designed program of prevention and support has the potential to maximize benefit and minimize harm. Conclusions Pediatric psychologists can play important roles in an era of expanded newborn screening by helping families understand genetic information, make informed decisions about genetic testing, and cope with the potential psychosocial consequences of genetic information.

Purpose This research provided a first-generation standardization of automated language environment estimates, validated these estimates against standard language assessments, and extended on previous research reporting language behavior differences across socioeconomic groups. Method Typically developing children between 2 to 48 months of age completed monthly, daylong recordings in their natural language environments over a span of approximately 6–38 months. The resulting data set contained 3,213 12-hr recordings automatically analyzed by using the Language Environment Analysis (LENA) System to generate estimates of (a) the number of adult words in the child's environment, (b) the amount of caregiver–child interaction, and (c) the frequency of child vocal output. Results Child vocalization frequency and turn-taking increased with age, whereas adult word counts were age independent after early infancy. Child vocalization and conversational turn estimates predicted 7%–16% of the variance observed in child language assessment scores. Lower socioeconomic status (SES) children produced fewer vocalizations, engaged in fewer adult–child interactions, and were exposed to fewer daily adult words compared with their higher socioeconomic status peers, but within-group variability was high. Conclusions The results offer new insight into the landscape of the early language environment, with clinical implications for identification of children at-risk for impoverished language environments.

Purpose To evaluate the efficacy of a 6-month course of responsivity education/prelinguistic milieu teaching (RE/PMT) for children with developmental delay and RE/PMT’s effects on parenting stress in a randomized clinical trial. Method Fifty-one children, age 24–33 months, with no more than 10 expressive words or signs, were randomly assigned to treatment/no-treatment groups. Thirteen children in each group had a diagnosis of Down syndrome. Results In 1 of 2 multivariate comparisons, the RE/PMT group exhibited superior gains in communication compared with the no-treatment group. The treatment effect for overall use of intentional communication acts in the child–examiner context was significant ( d = .68, 95% confidence interval = 0.12–1.24). There were no effects on child outcomes due to presence or absence of Down syndrome. RE/PMT led to modest increases in recoding of child acts by parents of children who did not have Down syndrome. There were no effects on parenting stress associated with the intervention or the presence or absence of Down syndrome. Conclusions RE/PMT may be applied clinically with the expectation of medium-size effects on the child’s rate of intentional communication acts after 6 months of intervention. The approach warrants further investigation with modifications, such as delivery at higher intensity levels.

A fundamental tenet of newborn screening is that screening should lead to a proven benefit for the infant. The standard is usually construed as medical benefit that significantly improves a child’s health. Screening for many conditions that cause developmental disabilities does not currently meet this standard. We argue for expanding concepts of presumptive benefit. Newborn screening provides access to early intervention programs that are shown to positively influence child development and support families. Consumers want information about their children’s health and their own reproductive risk, and they have a broader view than policymakers of what constitutes a treatable disorder. Newborn screening provides other societal benefits that, in the absence of data showing harm and with appropriate attention to ethical and legal issues, warrant consideration of an expansion of targets for newborn screening.

Language deficits occur in a variety of developmental disorders including autism spectrum disorders, Down syndrome, fragile X syndrome, specific language impairment, and Williams syndrome. This paper describes the specific pattern of linguistic deficits in each of these disorders in terms of speech production, semantic, and syntactic abilities as well as the relationship between cognitive and linguistic skills and the presence of a deviant or delayed pattern of development. In the spirit of synthesis across diverse literatures, preliminary comparisons among the language profiles of these disorders are made. The full picture, however, is incomplete given the current state of the literature, which tends to focus on the analysis of a narrow range of linguistic phenomena within a single disorder. The field is in need of research that systematically compares these disorders and leads to detailed descriptions of linguistic phenotypes of each disorder. The study of children with language disorders has a long and rich history. Language disorders are widely recognized as hallmark symptoms of an array of developmental clinical conditions. The research literature is generally organized according to diagnostic category, usually with the aim of describing the language disorders of a particular clinical group, for example, autism spectrum disorders, Down syndrome, fragile X syndrome, specific language impairment (SLI), or Williams syndrome. There are fewer investigations involving pairwise comparisons across conditions to determine if language and/or cognitive profiles are the same or different across clinical groups. Rice and Warren (2004) argue that there is a current need for careful consideration of the ways in which language disorders are manifest across clinical conditions, given the momentum in genetic and biobehavioral

The nature and causes of language disorders are research topics familiar to the readership ofApplied Psycholinguistics. Current inquiries place these topics in a dynamic and sprawling multidisciplinary context manifested through investigations of linguistic acquisition, cognitive development, genetics, neurocortical processes, cognitive neurolinguistics, behavioral phenotypes, and language intervention. This list is not complete by any means, but it does suggest the wide front of the current search for better knowledge about what causes language disorders and the dimensions of language acquisition that are affected. Although there is considerable momentum underway in current inquiries, the full potential for scientific advancement is hampered by fragmentation in the field, in part attributable to partitioning by diagnostic categories of affectedness. Investigators (and their funding sources) often focus on a particular clinical group, such as specific language impairment (SLI), autism/autism spectrum disorders (ASD), Williams syndrome (WS), Down syndrome (DS), or fragile X syndrome (FXS). Although this is not exclusively the case, it is relatively difficult to carry out comparative studies across the multiple clinical conditions in which language disorders appear and to monitor developments across such a wide front of investigation. The consequence is that findings are distributed across different publication outlets and different groups of scholars, a situation that can limit our appreciation of the ways in which language disorders are manifest, and the identification of common etiological factors.

The purpose of this study was to determine the extent to which naturalistic practices were being implemented across a representative sample of 17 early intervention programs and classroom activities. Measures of teacher-child communicative interactions, program quality, specialized training, and ongoing support were administered. Results indicated that teachers use descriptive talk more frequently than direct teaching strategies, particularly during freeplay activities. Implementation of direct teaching strategies was positively related to program quality, and specialized training might influence teacher's perspectives about use of freeplay as an opportunity to facilitate communication development. Data obtained in separate geographic regions of the country were remarkably similar, providing modest support for the external validity of these findings. Implications for practice and research are discussed.

Purpose In this study, we examine joint engagement (JE) in young children with fragile X syndrome (FXS) and its relationship to language abilities and autism spectrum disorder symptomatology at 24 to 36 months (toddler period) and 59 to 68 months (child period). Method Participants were 28 children with FXS (24 boys, four girls) and their mothers. Videotaped home observations were conducted during the toddler period and coded for JE. Language abilities were measured at both ages from a developmental assessment, a functional measure, and from a language sample. The Childhood Autism Rating Scale (Schopler, Reichler, & Renner, 1988) was completed at both ages. Results Children with FXS spent more time in supported JE than in coordinated JE. Using a weighted JE variable, we found that children with FXS who had higher weighted JE scores also had more advanced expressive language skills at both the toddler and child periods. Weighted JE was negatively related to autism symptomatology in the toddler period. Conclusion This study provides evidence that children with FXS who use more JE also have more advanced expressive language skills in early development. Therefore, existing early interventions that target JE behaviors may be effective for promoting language, social communication, and social interaction in this population. Supplemental Material https://doi.org/10.23641/asha.14963901

Purpose The authors examined (a) whether dose frequency of milieu communication teaching (MCT) affects children's canonical syllabic communication and (b) whether the relation between early canonical syllabic communication and later spoken vocabulary is mediated by parental linguistic mapping in children with intellectual disabilities (ID). Method The authors drew on extant data from a recent differential treatment intensity study in which 63 toddlers with ID were randomly assigned to receive either five 1-hr MCT sessions per week (i.e., daily treatment) or one 1-hr MCT session per week (i.e., weekly treatment) for 9 months. Children's early canonical syllabic communication was measured after 3 months of treatment, and later spoken vocabulary was measured at posttreatment. Midpoint parental linguistic mapping was measured after 6 months of treatment. Results A moderate-sized effect in favor of daily treatment was observed on canonical syllabic communication. The significant relation between canonical syllabic communication and spoken vocabulary was partially mediated by linguistic mapping. Conclusions These results suggest that canonical syllabic communication may elicit parental linguistic mapping, which may in turn support spoken vocabulary development in children with ID. More frequent early intervention boosted canonical syllabic communication, which may jump-start this transactional language-learning mechanism. Implications for theory, research, and practice are discussed.