Acoustic properties of early vocalizations in infants with fragile X syndrome / Lisa R. HAMRICK in Autism Research, 12-11 (November 2019)  

Public ISBD [article]  inAutism Research > 12-11 (November 2019) . - p.1663-1679 Titre : Acoustic properties of early vocalizations in infants with fragile X syndrome Type de document : texte imprimé Auteurs : Lisa R. HAMRICK, Auteur ; Amanda SEIDL, Auteur ; Bridgette L. TONNSEN, Auteur Article en page(s) : p.1663-1679 Langues : Anglais (eng) Mots-clés : acoustics  autism spectrum disorder  canonical babbling  fragile X syndrome  pitch  vocalization duration  volubility Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is a neurogenetic syndrome characterized by cognitive impairments and high rates of autism spectrum disorder (ASD). FXS is often highlighted as a model for exploring pathways of symptom expression in ASD due to the high prevalence of ASD symptoms in this population and the known single-gene cause of FXS. Early vocalization features-including volubility, complexity, duration, and pitch-have shown promise in detecting ASD in idiopathic ASD populations but have yet to be extensively studied in a population with a known genetic cause for ASD such as FXS. Investigating early trajectories of these features in FXS may inform our limited knowledge of potential mechanisms that predict later social communication outcomes. The present study addresses this need by presenting preliminary findings which (a) characterize early vocalization features in FXS relative to low-risk controls (LRC) and (b) test the specificity of associations between these features and language and ASD outcomes. We coded vocalization features during a standardized child-examiner interaction for 39 nine-month-olds (22 FXS, 17 LRC) whose clinical outcomes were assessed at 24 months. Our results provide preliminary evidence that within FXS, associations between vocalization features and 24-month language outcomes may diverge from those observed in LRC, and that vocalization features may be associated with later ASD symptoms. These findings provide a starting point for more research exploring these features as potential early markers of ASD in FXS, which in turn may lead to improved early identification methods, treatment approaches, and overall well-being of individuals with ASD. Autism Res2019. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Although vocal features of 9-month-olds with FXS did not differ from those of low-risk controls, several features were associated with later language and ASD outcomes at 24 months in FXS. These preliminary results suggest acoustic data may be related to clinical outcomes in FXS and potentially other high-risk populations. Further characterizing these associations may facilitate understanding of biological mechanisms and risk factors associated with social communication development and ASD. En ligne : http://dx.doi.org/10.1002/aur.2176 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=412 [article] Acoustic properties of early vocalizations in infants with fragile X syndrome [texte imprimé] / Lisa R. HAMRICK, Auteur ; Amanda SEIDL, Auteur ; Bridgette L. TONNSEN, Auteur . - p.1663-1679. Langues : Anglais (eng) in Autism Research > 12-11 (November 2019) . - p.1663-1679 Mots-clés : acoustics  autism spectrum disorder  canonical babbling  fragile X syndrome  pitch  vocalization duration  volubility Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is a neurogenetic syndrome characterized by cognitive impairments and high rates of autism spectrum disorder (ASD). FXS is often highlighted as a model for exploring pathways of symptom expression in ASD due to the high prevalence of ASD symptoms in this population and the known single-gene cause of FXS. Early vocalization features-including volubility, complexity, duration, and pitch-have shown promise in detecting ASD in idiopathic ASD populations but have yet to be extensively studied in a population with a known genetic cause for ASD such as FXS. Investigating early trajectories of these features in FXS may inform our limited knowledge of potential mechanisms that predict later social communication outcomes. The present study addresses this need by presenting preliminary findings which (a) characterize early vocalization features in FXS relative to low-risk controls (LRC) and (b) test the specificity of associations between these features and language and ASD outcomes. We coded vocalization features during a standardized child-examiner interaction for 39 nine-month-olds (22 FXS, 17 LRC) whose clinical outcomes were assessed at 24 months. Our results provide preliminary evidence that within FXS, associations between vocalization features and 24-month language outcomes may diverge from those observed in LRC, and that vocalization features may be associated with later ASD symptoms. These findings provide a starting point for more research exploring these features as potential early markers of ASD in FXS, which in turn may lead to improved early identification methods, treatment approaches, and overall well-being of individuals with ASD. Autism Res2019. (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: Although vocal features of 9-month-olds with FXS did not differ from those of low-risk controls, several features were associated with later language and ASD outcomes at 24 months in FXS. These preliminary results suggest acoustic data may be related to clinical outcomes in FXS and potentially other high-risk populations. Further characterizing these associations may facilitate understanding of biological mechanisms and risk factors associated with social communication development and ASD. En ligne : http://dx.doi.org/10.1002/aur.2176 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=412

Behavioral Markers of Emergent Stranger Anxiety in Infants and Toddlers with Fragile X Syndrome / Bridgette TONNSEN in Journal of Autism and Developmental Disorders, 47-11 (November 2017)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 47-11 (November 2017) . - p.3646-3658 Titre : Behavioral Markers of Emergent Stranger Anxiety in Infants and Toddlers with Fragile X Syndrome Type de document : texte imprimé Auteurs : Bridgette TONNSEN, Auteur ; Jessica F. SCHERR, Auteur ; Debra REISINGER, Auteur ; Jacqueline ROBERTS, Auteur Article en page(s) : p.3646-3658 Langues : Anglais (eng) Mots-clés : Anxiety  Autism  Behavior  Fragile X syndrome  Longitudinal  Social approach Index. décimale : PER Périodiques Résumé : Studying anxiety in neurogenetic syndromes may inform the intersection of biological and developmental risks, facilitating effective and targeted interventions. We longitudinally examined stranger fear in infants and toddlers with fragile X syndrome (FXS; n = 46) and typical controls (n = 33), as well as associations between observed stranger fear and rating scales of anxiety, withdrawal and autism features within FXS. Results indicated atypical facial fear in FXS, although facial fear did not index anxiety, autistic symptoms or social withdrawal. Instead, lower withdrawal was associated with decreased distress vocalizations across age, and higher autistic symptoms were associated with lower intensity escape behaviors. Early stranger fear in FXS reflects both typical and atypical dimensions and may help index emergence of social anxiety in this population. En ligne : http://dx.doi.org/10.1007/s10803-017-3270-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=325 [article] Behavioral Markers of Emergent Stranger Anxiety in Infants and Toddlers with Fragile X Syndrome [texte imprimé] / Bridgette TONNSEN, Auteur ; Jessica F. SCHERR, Auteur ; Debra REISINGER, Auteur ; Jacqueline ROBERTS, Auteur . - p.3646-3658. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 47-11 (November 2017) . - p.3646-3658 Mots-clés : Anxiety  Autism  Behavior  Fragile X syndrome  Longitudinal  Social approach Index. décimale : PER Périodiques Résumé : Studying anxiety in neurogenetic syndromes may inform the intersection of biological and developmental risks, facilitating effective and targeted interventions. We longitudinally examined stranger fear in infants and toddlers with fragile X syndrome (FXS; n = 46) and typical controls (n = 33), as well as associations between observed stranger fear and rating scales of anxiety, withdrawal and autism features within FXS. Results indicated atypical facial fear in FXS, although facial fear did not index anxiety, autistic symptoms or social withdrawal. Instead, lower withdrawal was associated with decreased distress vocalizations across age, and higher autistic symptoms were associated with lower intensity escape behaviors. Early stranger fear in FXS reflects both typical and atypical dimensions and may help index emergence of social anxiety in this population. En ligne : http://dx.doi.org/10.1007/s10803-017-3270-6 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=325

Brief Report: Autism Symptoms in Infants with Fragile X Syndrome / Jane E. ROBERTS in Journal of Autism and Developmental Disorders, 46-12 (December 2016)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 46-12 (December 2016) . - p.3830-3837 Titre : Brief Report: Autism Symptoms in Infants with Fragile X Syndrome Type de document : texte imprimé Auteurs : Jane E. ROBERTS, Auteur ; Bridgette L. TONNSEN, Auteur ; Lindsay M. MCCARY, Auteur ; Kelly E. CARAVELLA, Auteur ; Svetlana V. SHINKAREVA, Auteur Article en page(s) : p.3830-3837 Langues : Anglais (eng) Mots-clés : Autism  Fragile X  Infants  Autism Observation Scale for Infants Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is the most common known genetic cause of autism spectrum disorder (ASD). Although 50–75 % of children with FXS meet ASD criteria, no studies have compared ASD symptoms in infants with FXS versus other high risk groups, such as siblings of children with ASD (ASIBs). Using the Autism Observation Scale for Infants, our findings indicate that 53 % of 12-month infants with FXS fall in the “at risk” category compared to 17 and 6 % for age-matched ASIBs and controls, respectively. Elevated atypical motor behaviors were associated with elevated risk for FXS. Cross-syndrome comparisons are essential to understanding the heterogeneity of ASD and identifying candidate markers that will facilitate differential diagnosis of ASD in genetic disorders such as FXS. En ligne : http://dx.doi.org/10.1007/s10803-016-2903-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=297 [article] Brief Report: Autism Symptoms in Infants with Fragile X Syndrome [texte imprimé] / Jane E. ROBERTS, Auteur ; Bridgette L. TONNSEN, Auteur ; Lindsay M. MCCARY, Auteur ; Kelly E. CARAVELLA, Auteur ; Svetlana V. SHINKAREVA, Auteur . - p.3830-3837. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 46-12 (December 2016) . - p.3830-3837 Mots-clés : Autism  Fragile X  Infants  Autism Observation Scale for Infants Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is the most common known genetic cause of autism spectrum disorder (ASD). Although 50–75 % of children with FXS meet ASD criteria, no studies have compared ASD symptoms in infants with FXS versus other high risk groups, such as siblings of children with ASD (ASIBs). Using the Autism Observation Scale for Infants, our findings indicate that 53 % of 12-month infants with FXS fall in the “at risk” category compared to 17 and 6 % for age-matched ASIBs and controls, respectively. Elevated atypical motor behaviors were associated with elevated risk for FXS. Cross-syndrome comparisons are essential to understanding the heterogeneity of ASD and identifying candidate markers that will facilitate differential diagnosis of ASD in genetic disorders such as FXS. En ligne : http://dx.doi.org/10.1007/s10803-016-2903-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=297

Brief Report: Challenging Behaviors in Toddlers and Preschoolers with Angelman, Prader-Willi, and Williams Syndromes / Wei Siong NEO in Journal of Autism and Developmental Disorders, 49-4 (April 2019)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 49-4 (April 2019) . - p.1717-1726 Titre : Brief Report: Challenging Behaviors in Toddlers and Preschoolers with Angelman, Prader-Willi, and Williams Syndromes Type de document : texte imprimé Auteurs : Wei Siong NEO, Auteur ; Bridgette L. TONNSEN, Auteur Article en page(s) : p.1717-1726 Langues : Anglais (eng) Mots-clés : Angelman syndrome  Challenging behavior  Child Behavior Checklist  Early childhood  Prader-Willi syndrome  Williams syndrome Index. décimale : PER Périodiques Résumé : Children with neurogenetic syndromes (NGS) experience comorbid challenging behaviors and psychopathology. We examined challenging behaviors in 86 toddlers and preschoolers across three NGS [Angelman syndrome (AS), Prader-Willi syndrome (PWS), and Williams syndrome (WS)] and 43 low-risk controls (LRC), using the Child Behavior Checklist for Ages 1(1/2)-5. Challenging behavior profiles differed across NGS, with generally elevated behaviors in AS and WS, but not PWS, relative to LRC. Withdrawn and autism spectrum symptoms were particularly elevated in AS. Although several profiles were similar to those previously reported in older children and adults, we also observed inconsistencies that suggest non-linear developmental patterns of challenging behaviors. These findings underscore the importance of characterizing early challenging behaviors to inform atypical phenotypic development and targeted intervention. En ligne : https://dx.doi.org/10.1007/s10803-018-3853-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=388 [article] Brief Report: Challenging Behaviors in Toddlers and Preschoolers with Angelman, Prader-Willi, and Williams Syndromes [texte imprimé] / Wei Siong NEO, Auteur ; Bridgette L. TONNSEN, Auteur . - p.1717-1726. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 49-4 (April 2019) . - p.1717-1726 Mots-clés : Angelman syndrome  Challenging behavior  Child Behavior Checklist  Early childhood  Prader-Willi syndrome  Williams syndrome Index. décimale : PER Périodiques Résumé : Children with neurogenetic syndromes (NGS) experience comorbid challenging behaviors and psychopathology. We examined challenging behaviors in 86 toddlers and preschoolers across three NGS [Angelman syndrome (AS), Prader-Willi syndrome (PWS), and Williams syndrome (WS)] and 43 low-risk controls (LRC), using the Child Behavior Checklist for Ages 1(1/2)-5. Challenging behavior profiles differed across NGS, with generally elevated behaviors in AS and WS, but not PWS, relative to LRC. Withdrawn and autism spectrum symptoms were particularly elevated in AS. Although several profiles were similar to those previously reported in older children and adults, we also observed inconsistencies that suggest non-linear developmental patterns of challenging behaviors. These findings underscore the importance of characterizing early challenging behaviors to inform atypical phenotypic development and targeted intervention. En ligne : https://dx.doi.org/10.1007/s10803-018-3853-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=388

Dyadic interactions in children exhibiting the broader autism phenotype: Is the broader autism phenotype distinguishable from typical development? / Ashleigh M. KELLERMAN in Autism Research, 12-3 (March 2019)  

Public ISBD [article]  inAutism Research > 12-3 (March 2019) . - p.469-481 Titre : Dyadic interactions in children exhibiting the broader autism phenotype: Is the broader autism phenotype distinguishable from typical development? Type de document : texte imprimé Auteurs : Ashleigh M. KELLERMAN, Auteur ; A.J. SCHWICHTENBERG, Auteur ; Bridgette L. TONNSEN, Auteur ; G. POSADA, Auteur ; Sean P. LANE, Auteur Article en page(s) : p.469-481 Langues : Anglais (eng) Mots-clés : autism spectrum disorder  broader autism phenotype  infant sibling  joint engagement  mother-child interactions  responsiveness Index. décimale : PER Périodiques Résumé : In families raising a child with an autism spectrum disorder (ASD), infant siblings are at elevated risk for ASD and other developmental concerns, including elements of the broader autism phenotype (BAP). Typically, the BAP is indexed using standardized developmental assessments; however, these measures do not capture a number of social difficulties commonly associated with the BAP. The present study aims to expand our developmental understanding of the BAP by comparing children exhibiting the BAP to their typically developing peers on, (a) standardized measures of development, and (b) social behaviors exhibited during dyadic play interactions. As part of a prospective study, dyads were recruited from families with at least one older child with ASD (high-risk, n = 36), and families with no history of ASD (low-risk, n = 38). During laboratory visits at 12, 15, 18, and 24 months of age, infants completed a series of standardized assessments and a mother-child play interaction. Dyadic play interactions were micro-analytically coded for gaze, positive affect, and vocalizations to create theory-driven composites to index dyadic synchrony and responsiveness. Videos were also coded with an existing rating scheme for joint engagement and child responsiveness. Multilevel models revealed significant group differences on select constructs within the first 2 years. Language and cognitive differences emerged by 24 months of age, whereas dyadic differences were evident as early as 15 months. Recognizing the increasing demand for elevated-risk interventions, these findings highlight several social constructs through which interventions may identify risk and promote optimal development. Autism Res 2019, 12: 469-481 (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: In families raising children with an autism spectrum disorder (ASD), younger siblings are at an increased risk for social and developmental difficulties that characterize a "broader autism phenotype." The present study explored the emergence of social, language, and cognitive differences in the first 2 years of life. Social differences were evident as early as 15 months of age for several play-based measures, and language and cognitive differences emerged by 24 months of age. For infant siblings of children with ASD, some of the earliest behavioral marks for subclinical features of ASD are evident within the first 2 years of life. En ligne : http://dx.doi.org/10.1002/aur.2062 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=387 [article] Dyadic interactions in children exhibiting the broader autism phenotype: Is the broader autism phenotype distinguishable from typical development? [texte imprimé] / Ashleigh M. KELLERMAN, Auteur ; A.J. SCHWICHTENBERG, Auteur ; Bridgette L. TONNSEN, Auteur ; G. POSADA, Auteur ; Sean P. LANE, Auteur . - p.469-481. Langues : Anglais (eng) in Autism Research > 12-3 (March 2019) . - p.469-481 Mots-clés : autism spectrum disorder  broader autism phenotype  infant sibling  joint engagement  mother-child interactions  responsiveness Index. décimale : PER Périodiques Résumé : In families raising a child with an autism spectrum disorder (ASD), infant siblings are at elevated risk for ASD and other developmental concerns, including elements of the broader autism phenotype (BAP). Typically, the BAP is indexed using standardized developmental assessments; however, these measures do not capture a number of social difficulties commonly associated with the BAP. The present study aims to expand our developmental understanding of the BAP by comparing children exhibiting the BAP to their typically developing peers on, (a) standardized measures of development, and (b) social behaviors exhibited during dyadic play interactions. As part of a prospective study, dyads were recruited from families with at least one older child with ASD (high-risk, n = 36), and families with no history of ASD (low-risk, n = 38). During laboratory visits at 12, 15, 18, and 24 months of age, infants completed a series of standardized assessments and a mother-child play interaction. Dyadic play interactions were micro-analytically coded for gaze, positive affect, and vocalizations to create theory-driven composites to index dyadic synchrony and responsiveness. Videos were also coded with an existing rating scheme for joint engagement and child responsiveness. Multilevel models revealed significant group differences on select constructs within the first 2 years. Language and cognitive differences emerged by 24 months of age, whereas dyadic differences were evident as early as 15 months. Recognizing the increasing demand for elevated-risk interventions, these findings highlight several social constructs through which interventions may identify risk and promote optimal development. Autism Res 2019, 12: 469-481 (c) 2019 International Society for Autism Research, Wiley Periodicals, Inc. LAY SUMMARY: In families raising children with an autism spectrum disorder (ASD), younger siblings are at an increased risk for social and developmental difficulties that characterize a "broader autism phenotype." The present study explored the emergence of social, language, and cognitive differences in the first 2 years of life. Social differences were evident as early as 15 months of age for several play-based measures, and language and cognitive differences emerged by 24 months of age. For infant siblings of children with ASD, some of the earliest behavioral marks for subclinical features of ASD are evident within the first 2 years of life. En ligne : http://dx.doi.org/10.1002/aur.2062 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=387

Heart rate-defined sustained attention in infants at risk for autism / Bridgette L. TONNSEN in Journal of Neurodevelopmental Disorders, 10-1 (December 2018)  

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Middle School Students’ Attitudes Toward a Peer With Autism Spectrum Disorder / Bridgette L. TONNSEN in Focus on Autism and Other Developmental Disabilities, 31-4 (December 2016)  

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Social Avoidance Emerges in Infancy and Persists into Adulthood in Fragile X Syndrome / Jacqueline ROBERTS in Journal of Autism and Developmental Disorders, 49-9 (September 2019)  

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Treatment effects of stimulant medication in young boys with fragile X syndrome / Jane E. ROBERTS in Journal of Neurodevelopmental Disorders, 3-3 (September 2011)  

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