Developmental associations between motor and communication outcomes in Fragile X syndrome: Variation in the context of co-occurring autism / Kimberly HILLS ; Kayla SMITH ; Samuel MCQUILLIN ; Jane E. ROBERTS in Autism, 28-9 (September 2024)  

Public ISBD [article]  inAutism > 28-9 (September 2024) . - p.2191-2203 Titre : Developmental associations between motor and communication outcomes in Fragile X syndrome: Variation in the context of co-occurring autism Type de document : texte imprimé Auteurs : Kimberly HILLS, Auteur ; Kayla SMITH, Auteur ; Samuel MCQUILLIN, Auteur ; Jane E. ROBERTS, Auteur Article en page(s) : p.2191-2203 Langues : (en) Mots-clés : autism spectrum disorder  communication  development  Fragile X syndrome  motor development Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS), the leading heritable cause of intellectual disability, has a co-occurrence rate of autism spectrum disorder (ASD) estimated at ~60%. The onset and rates of motor development in FXS are slower relative to neurotypical development, and even more so in the context of co-occurring FXS+ASD. Extant evidence suggests these differences are likely to affect communication, yet this developmental process or how it varies in the context of co-occurring ASD remains unknown in FXS. We aimed to delineate developmental associations between early motor abilities and their rate of development from 9 to 60 months of age on communication outcomes in 51 children with FXS, 28 of whom had co-occurring ASD. We also aimed to identify variation in these developmental associations in the context of co-occurring ASD. Results captured within-syndrome variability in these developmental associations as a function of co-occurring ASD. Fine motor proved to be a robust predictor of receptive communication regardless of co-occurring ASD, but we identified differences between FXS with and without ASD in the association between aspects of motor development and expressive outcomes. Findings provide evidence for differential developmental processes in the context of co-occurring ASD with implications for timely developmental intervention. Lay abstract Fragile X syndrome (FXS), the leading heritable cause of intellectual disability, has a co-occurrence rate of autism spectrum disorder (ASD) estimated at ~60%. Children with FXS experience delayed achievement and slower development of key motor abilities, which happens to an even greater extent for children with both FXS and ASD. A multitude of studies have demonstrated that motor abilities are foundational skills related to later communication outcomes in neurotypical development, as well as in the context of ASD. However, these associations remain unexamined in FXS, or FXS+ASD. In this study, we aimed to determine the associations between early motor skills and their rate of development on communication outcomes in FXS. Furthermore, we investigated whether these associations varied in the context of co-occurring FXS+ASD. Results revealed within-FXS variation in the context of co-occurring ASD between some aspects of motor development and communication outcomes, yet within-FXS consistency between others. Findings provide evidence for variability in developmental processes and outcomes in FXS in the context of co-occurring ASD and offer implications for intervention. En ligne : https://dx.doi.org/10.1177/13623613231225498 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=535 [article] Developmental associations between motor and communication outcomes in Fragile X syndrome: Variation in the context of co-occurring autism [texte imprimé] / Kimberly HILLS, Auteur ; Kayla SMITH, Auteur ; Samuel MCQUILLIN, Auteur ; Jane E. ROBERTS, Auteur . - p.2191-2203. Langues : (en) in Autism > 28-9 (September 2024) . - p.2191-2203 Mots-clés : autism spectrum disorder  communication  development  Fragile X syndrome  motor development Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS), the leading heritable cause of intellectual disability, has a co-occurrence rate of autism spectrum disorder (ASD) estimated at ~60%. The onset and rates of motor development in FXS are slower relative to neurotypical development, and even more so in the context of co-occurring FXS+ASD. Extant evidence suggests these differences are likely to affect communication, yet this developmental process or how it varies in the context of co-occurring ASD remains unknown in FXS. We aimed to delineate developmental associations between early motor abilities and their rate of development from 9 to 60 months of age on communication outcomes in 51 children with FXS, 28 of whom had co-occurring ASD. We also aimed to identify variation in these developmental associations in the context of co-occurring ASD. Results captured within-syndrome variability in these developmental associations as a function of co-occurring ASD. Fine motor proved to be a robust predictor of receptive communication regardless of co-occurring ASD, but we identified differences between FXS with and without ASD in the association between aspects of motor development and expressive outcomes. Findings provide evidence for differential developmental processes in the context of co-occurring ASD with implications for timely developmental intervention. Lay abstract Fragile X syndrome (FXS), the leading heritable cause of intellectual disability, has a co-occurrence rate of autism spectrum disorder (ASD) estimated at ~60%. Children with FXS experience delayed achievement and slower development of key motor abilities, which happens to an even greater extent for children with both FXS and ASD. A multitude of studies have demonstrated that motor abilities are foundational skills related to later communication outcomes in neurotypical development, as well as in the context of ASD. However, these associations remain unexamined in FXS, or FXS+ASD. In this study, we aimed to determine the associations between early motor skills and their rate of development on communication outcomes in FXS. Furthermore, we investigated whether these associations varied in the context of co-occurring FXS+ASD. Results revealed within-FXS variation in the context of co-occurring ASD between some aspects of motor development and communication outcomes, yet within-FXS consistency between others. Findings provide evidence for variability in developmental processes and outcomes in FXS in the context of co-occurring ASD and offer implications for intervention. En ligne : https://dx.doi.org/10.1177/13623613231225498 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=535

Early negative affect in males and females with fragile X syndrome: implications for anxiety and autism / Carla A. WALL in Journal of Neurodevelopmental Disorders, 11-1 (December 2019)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 11-1 (December 2019) . - 22 p. Titre : Early negative affect in males and females with fragile X syndrome: implications for anxiety and autism Type de document : texte imprimé Auteurs : Carla A. WALL, Auteur ; Abigail L. HOGAN-BROWN, Auteur ; Elizabeth A. WILL, Auteur ; Samuel MCQUILLIN, Auteur ; Bridgette L. KELLEHER, Auteur ; Jane E. ROBERTS, Auteur Article en page(s) : 22 p. Langues : Anglais (eng) Mots-clés : Anxiety  Autism spectrum disorder  Fragile X syndrome  Negative affect  Sex differences Index. décimale : PER Périodiques Résumé : BACKGROUND: Fragile X syndrome (FXS) is a genetic disorder that is highly comorbid with anxiety and autism spectrum disorder (ASD). Elevated negative affect in young children has been associated with increased risk for both anxiety and ASD; however, these relations remain poorly understood in FXS. METHODS: The present prospective longitudinal study examined the trajectory of negative affect from infancy through preschool in males and females with FXS and typical development and its relation to anxiety and ASD. RESULTS: Results indicate a complex association reflecting group, developmental, and sex effects. Specifically, the group with FXS displayed a trajectory of increasing negative affect across age that was distinct from the typical controls. This atypical trajectory of negative affect in FXS was driven by sex effects in that males showed lower negative affect during infancy followed by steep increases across the toddler and preschool years whereas the females displayed a flatter trajectory. Finally, elevated negative affect predicted anxiety symptoms in males, but not females, with no relationship to ASD in males or females with FXS. CONCLUSIONS: The current work addresses the importance of studying the development of psychopathology in a specific neurogenetic population. Temperamental negative affect was shown to be an important early marker for anxiety in young children with FXS, with subtle differences observed between males and females. En ligne : https://dx.doi.org/10.1186/s11689-019-9284-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=409 [article] Early negative affect in males and females with fragile X syndrome: implications for anxiety and autism [texte imprimé] / Carla A. WALL, Auteur ; Abigail L. HOGAN-BROWN, Auteur ; Elizabeth A. WILL, Auteur ; Samuel MCQUILLIN, Auteur ; Bridgette L. KELLEHER, Auteur ; Jane E. ROBERTS, Auteur . - 22 p. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 11-1 (December 2019) . - 22 p. Mots-clés : Anxiety  Autism spectrum disorder  Fragile X syndrome  Negative affect  Sex differences Index. décimale : PER Périodiques Résumé : BACKGROUND: Fragile X syndrome (FXS) is a genetic disorder that is highly comorbid with anxiety and autism spectrum disorder (ASD). Elevated negative affect in young children has been associated with increased risk for both anxiety and ASD; however, these relations remain poorly understood in FXS. METHODS: The present prospective longitudinal study examined the trajectory of negative affect from infancy through preschool in males and females with FXS and typical development and its relation to anxiety and ASD. RESULTS: Results indicate a complex association reflecting group, developmental, and sex effects. Specifically, the group with FXS displayed a trajectory of increasing negative affect across age that was distinct from the typical controls. This atypical trajectory of negative affect in FXS was driven by sex effects in that males showed lower negative affect during infancy followed by steep increases across the toddler and preschool years whereas the females displayed a flatter trajectory. Finally, elevated negative affect predicted anxiety symptoms in males, but not females, with no relationship to ASD in males or females with FXS. CONCLUSIONS: The current work addresses the importance of studying the development of psychopathology in a specific neurogenetic population. Temperamental negative affect was shown to be an important early marker for anxiety in young children with FXS, with subtle differences observed between males and females. En ligne : https://dx.doi.org/10.1186/s11689-019-9284-y Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=409

Emergence and rate of autism in fragile X syndrome across the first years of life / Jane E. ROBERTS in Development and Psychopathology, 32-4 (October 2020)  

Public ISBD [article]  inDevelopment and Psychopathology > 32-4 (October 2020) . - p.1335-1352 Titre : Emergence and rate of autism in fragile X syndrome across the first years of life Type de document : texte imprimé Auteurs : Jane E. ROBERTS, Auteur ; Jessica BRADSHAW, Auteur ; Elizabeth A. WILL, Auteur ; Abigail L. HOGAN, Auteur ; Samuel MCQUILLIN, Auteur ; Kimberly HILLS, Auteur Article en page(s) : p.1335-1352 Langues : Anglais (eng) Mots-clés : autism spectrum disorder  developmental trajectory  early identification  fragile X syndrome  infants Index. décimale : PER Périodiques Résumé : Prospective longitudinal studies of idiopathic autism spectrum disorder (ASD) have provided insights into early symptoms and predictors of ASD during infancy, well before ASD can be diagnosed at age 2-3 years. However, research on the emergence of ASD in disorders with a known genetic etiology, contextualized in a developmental framework, is currently lacking. Using a biobehavioral multimethod approach, we (a) determined the rate of ASD in N = 51 preschoolers with fragile X syndrome (FXS) using a clinical best estimate (CBE) procedure with differential diagnoses of comorbid psychiatric disorders and (b) investigated trajectories of ASD symptoms and physiological arousal across infancy as predictors of ASD in preschoolers with FXS. ASD was not diagnosed if intellectual ability or psychiatric disorders better accounted for the symptoms. Our results determined that 60.7% of preschoolers with FXS met the Diagnostic and Statistical Manual of Mental Disorders (fifth edition) (DSM-5) criteria for ASD using the CBE procedure. In addition, 92% of these preschoolers presented with developmental delay and 45.4% also met criteria for psychiatric disorders, either anxiety, ADHD, or both. ASD diagnoses in preschoolers with FXS were predicted by elevated scores on traditional ASD screeners in addition to elevated autonomic arousal and avoidant eye contact from infancy. En ligne : http://dx.doi.org/10.1017/s0954579420000942 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=433 [article] Emergence and rate of autism in fragile X syndrome across the first years of life [texte imprimé] / Jane E. ROBERTS, Auteur ; Jessica BRADSHAW, Auteur ; Elizabeth A. WILL, Auteur ; Abigail L. HOGAN, Auteur ; Samuel MCQUILLIN, Auteur ; Kimberly HILLS, Auteur . - p.1335-1352. Langues : Anglais (eng) in Development and Psychopathology > 32-4 (October 2020) . - p.1335-1352 Mots-clés : autism spectrum disorder  developmental trajectory  early identification  fragile X syndrome  infants Index. décimale : PER Périodiques Résumé : Prospective longitudinal studies of idiopathic autism spectrum disorder (ASD) have provided insights into early symptoms and predictors of ASD during infancy, well before ASD can be diagnosed at age 2-3 years. However, research on the emergence of ASD in disorders with a known genetic etiology, contextualized in a developmental framework, is currently lacking. Using a biobehavioral multimethod approach, we (a) determined the rate of ASD in N = 51 preschoolers with fragile X syndrome (FXS) using a clinical best estimate (CBE) procedure with differential diagnoses of comorbid psychiatric disorders and (b) investigated trajectories of ASD symptoms and physiological arousal across infancy as predictors of ASD in preschoolers with FXS. ASD was not diagnosed if intellectual ability or psychiatric disorders better accounted for the symptoms. Our results determined that 60.7% of preschoolers with FXS met the Diagnostic and Statistical Manual of Mental Disorders (fifth edition) (DSM-5) criteria for ASD using the CBE procedure. In addition, 92% of these preschoolers presented with developmental delay and 45.4% also met criteria for psychiatric disorders, either anxiety, ADHD, or both. ASD diagnoses in preschoolers with FXS were predicted by elevated scores on traditional ASD screeners in addition to elevated autonomic arousal and avoidant eye contact from infancy. En ligne : http://dx.doi.org/10.1017/s0954579420000942 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=433

Understanding perceptions underlying the self-reported stress among parents of adolescents with autism spectrum disorder: Considerations for supporting families / Natalie S. RAFF in Research in Autism Spectrum Disorders, 84 (June 2021)  

Public ISBD [article]  inResearch in Autism Spectrum Disorders > 84 (June 2021) . - 101770 Titre : Understanding perceptions underlying the self-reported stress among parents of adolescents with autism spectrum disorder: Considerations for supporting families Type de document : texte imprimé Auteurs : Natalie S. RAFF, Auteur ; Sarah S. MIRE, Auteur ; Leslie FRANKEL, Auteur ; Samuel D. MCQUILLIN, Auteur ; Katherine A. LOVELAND, Auteur ; Andrew DAIRE, Auteur ; Stacey C. GREBE, Auteur ; Georgina ROSENBROCK, Auteur Article en page(s) : 101770 Langues : Anglais (eng) Mots-clés : Adolescents  Autism spectrum disorder  Family support  IPQ-R-ASD  Parenting stress Index. décimale : PER Périodiques Résumé : Background Parents raising adolescents with autism spectrum disorder (ASD) often report higher stress than other parents. The influence of parents’ internal, or cognitive, experiences (i.e., their own perceptions) on this elevated stress has yet to be explored. Addressing this gap may reveal opportunities for enhancing support for families by elucidating malleable targets for reducing parents’ self-reported stress and/or informing family-focused intervention. The Double ABCX Model of Family Adaptation is a framework for understanding how perceptions, social support, and personal resources (i.e., coping) may affect stress. Methods We examined parents’ perceptions about ASD, perceived support, and coping among 214 parents of adolescents with ASD. Regression analyses were used to explore whether these factors were associated with parenting stress among those raising adolescents with ASD. Moderation effects of positive coping on the relationship between parent perceptions and parenting stress were also explored. Results Parent perceptions about within family support, the extent of ASD symptom predictability, and treatment being able to ‘control’ ASD were related to parenting stress. However, positive coping did not moderate the relationship between these perceptions and parenting stress. Conclusions Study findings emphasize significant associations between specific parental perceptions and the self-reported stress among families of adolescents with ASD. Particularly important to parenting stress were how much parents’ perceived adequate support within the family, that treatment was useful for controlling their adolescent’s ASD, and that their adolescent’s symptoms were predictable. These findings suggest that the way parents think about their adolescents’ ASD and the adequacy of the support within their own families are associated with parenting stress, and therefore may serve as treatment targets for positively affecting whole family outcomes, as well as foundations for additional research. En ligne : https://doi.org/10.1016/j.rasd.2021.101770 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=446 [article] Understanding perceptions underlying the self-reported stress among parents of adolescents with autism spectrum disorder: Considerations for supporting families [texte imprimé] / Natalie S. RAFF, Auteur ; Sarah S. MIRE, Auteur ; Leslie FRANKEL, Auteur ; Samuel D. MCQUILLIN, Auteur ; Katherine A. LOVELAND, Auteur ; Andrew DAIRE, Auteur ; Stacey C. GREBE, Auteur ; Georgina ROSENBROCK, Auteur . - 101770. Langues : Anglais (eng) in Research in Autism Spectrum Disorders > 84 (June 2021) . - 101770 Mots-clés : Adolescents  Autism spectrum disorder  Family support  IPQ-R-ASD  Parenting stress Index. décimale : PER Périodiques Résumé : Background Parents raising adolescents with autism spectrum disorder (ASD) often report higher stress than other parents. The influence of parents’ internal, or cognitive, experiences (i.e., their own perceptions) on this elevated stress has yet to be explored. Addressing this gap may reveal opportunities for enhancing support for families by elucidating malleable targets for reducing parents’ self-reported stress and/or informing family-focused intervention. The Double ABCX Model of Family Adaptation is a framework for understanding how perceptions, social support, and personal resources (i.e., coping) may affect stress. Methods We examined parents’ perceptions about ASD, perceived support, and coping among 214 parents of adolescents with ASD. Regression analyses were used to explore whether these factors were associated with parenting stress among those raising adolescents with ASD. Moderation effects of positive coping on the relationship between parent perceptions and parenting stress were also explored. Results Parent perceptions about within family support, the extent of ASD symptom predictability, and treatment being able to ‘control’ ASD were related to parenting stress. However, positive coping did not moderate the relationship between these perceptions and parenting stress. Conclusions Study findings emphasize significant associations between specific parental perceptions and the self-reported stress among families of adolescents with ASD. Particularly important to parenting stress were how much parents’ perceived adequate support within the family, that treatment was useful for controlling their adolescent’s ASD, and that their adolescent’s symptoms were predictable. These findings suggest that the way parents think about their adolescents’ ASD and the adequacy of the support within their own families are associated with parenting stress, and therefore may serve as treatment targets for positively affecting whole family outcomes, as well as foundations for additional research. En ligne : https://doi.org/10.1016/j.rasd.2021.101770 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=446

Using latent class analysis to identify treatment-use subgroups among parents of children with autism spectrum disorder / Sarah S. MIRE in Autism Research, 12-5 (May 2019)  

Public ISBD [article]  inAutism Research > 12-5 (May 2019) . - p.843-854 Titre : Using latent class analysis to identify treatment-use subgroups among parents of children with autism spectrum disorder Type de document : texte imprimé Auteurs : Sarah S. MIRE, Auteur ; Samuel MCQUILLIN, Auteur ; Madeline RACINE, Auteur ; Robin P. GOIN-KOCHEL, Auteur Article en page(s) : p.843-854 Langues : Anglais (eng) Mots-clés : Asd  autism  developmental regression  latent class analysis  treatment Index. décimale : PER Périodiques Résumé : Among parents of 2,582 children (ages 4-17 years old) with autism spectrum disorder (ASD), we used latent class analysis to identify subgroups and profiles of treatment users and included annual household income in the specification of the models, then described characteristics of each subgroup. Based on three indicators of fit (Akaike's Information Criterion, Bayesian Information Criterion, and Lo-Mendell-Rubin), six latent classes of treatment users emerged. Subgroups included users of: (a) mostly private and school speech and occupational therapies; (b) nearly all treatment types; (c) mostly speech and occupational therapies, plus intensive behavioral and "other" treatments, but little medication use; (d) private therapies almost exclusively; (e) primarily psychotropic medications; and (f) mostly school-based therapies. Income significantly predicted class differences for all but one latent class. Probabilities of families' lifetime use of nine treatment types varied depending on latent classification. Proportions of families reporting having observed children's developmental regression were largest in those with the highest overall treatment use, and these children also had the lowest cognitive and adaptive-functioning scores and the highest ASD symptom scores. Understanding patterns of treatment use among families of children with ASD is an important first step in enhancing treatment-related selection and implementation. Autism Research 2019, 12: 843-854. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: We identified six different groups of treatment users to help explain patterns in treatment implementation among parents of children and adolescents with autism. These included families who used: (a) mostly used private and school speech and occupational therapies; (b) nearly all treatment types (private and school therapies, intensive behavioral, biomedical, psychotropic medications, and other treatments); (c) mostly speech and occupational therapies, plus intensive behavioral and "other" treatments, but little medication use; (d) private therapies almost exclusively; (e) primarily psychotropic medications; and (f) mostly school-based therapies. En ligne : http://dx.doi.org/10.1002/aur.2107 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=397 [article] Using latent class analysis to identify treatment-use subgroups among parents of children with autism spectrum disorder [texte imprimé] / Sarah S. MIRE, Auteur ; Samuel MCQUILLIN, Auteur ; Madeline RACINE, Auteur ; Robin P. GOIN-KOCHEL, Auteur . - p.843-854. Langues : Anglais (eng) in Autism Research > 12-5 (May 2019) . - p.843-854 Mots-clés : Asd  autism  developmental regression  latent class analysis  treatment Index. décimale : PER Périodiques Résumé : Among parents of 2,582 children (ages 4-17 years old) with autism spectrum disorder (ASD), we used latent class analysis to identify subgroups and profiles of treatment users and included annual household income in the specification of the models, then described characteristics of each subgroup. Based on three indicators of fit (Akaike's Information Criterion, Bayesian Information Criterion, and Lo-Mendell-Rubin), six latent classes of treatment users emerged. Subgroups included users of: (a) mostly private and school speech and occupational therapies; (b) nearly all treatment types; (c) mostly speech and occupational therapies, plus intensive behavioral and "other" treatments, but little medication use; (d) private therapies almost exclusively; (e) primarily psychotropic medications; and (f) mostly school-based therapies. Income significantly predicted class differences for all but one latent class. Probabilities of families' lifetime use of nine treatment types varied depending on latent classification. Proportions of families reporting having observed children's developmental regression were largest in those with the highest overall treatment use, and these children also had the lowest cognitive and adaptive-functioning scores and the highest ASD symptom scores. Understanding patterns of treatment use among families of children with ASD is an important first step in enhancing treatment-related selection and implementation. Autism Research 2019, 12: 843-854. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: We identified six different groups of treatment users to help explain patterns in treatment implementation among parents of children and adolescents with autism. These included families who used: (a) mostly used private and school speech and occupational therapies; (b) nearly all treatment types (private and school therapies, intensive behavioral, biomedical, psychotropic medications, and other treatments); (c) mostly speech and occupational therapies, plus intensive behavioral and "other" treatments, but little medication use; (d) private therapies almost exclusively; (e) primarily psychotropic medications; and (f) mostly school-based therapies. En ligne : http://dx.doi.org/10.1002/aur.2107 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=397

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