Anxiety disorders in children with williams syndrome, their mothers, and their siblings: implications for the etiology of anxiety disorders / O. LEYFER in Journal of Neurodevelopmental Disorders, 1-1 (March 2009)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 1-1 (March 2009) . - p.4-14 Titre : Anxiety disorders in children with williams syndrome, their mothers, and their siblings: implications for the etiology of anxiety disorders Type de document : Texte imprimé et/ou numérique Auteurs : O. LEYFER, Auteur ; J. WOODRUFF-BORDEN, Auteur ; C. B. MERVIS, Auteur Article en page(s) : p.4-14 Langues : Anglais (eng) Mots-clés : Anxiety  Family  Genetics  Intellectual disability  Mental retardation  Williams syndrome Index. décimale : PER Périodiques Résumé : PURPOSE: To determine the prevalence of anxiety disorders in children with Williams syndrome (WS), their sibling closest in age, and their mothers and to examine the predictors of anxiety in these groups. METHODS: The prevalence of anxiety disorders was assessed and compared to that in the general population. RESULTS: Children with WS had a significantly higher prevalence of specific phobia, generalized anxiety disorder (GAD), and separation anxiety in comparison to children in the general population. While mothers had a higher prevalence of GAD than population controls, the excess was accounted for by mothers who had onset after the birth of their WS child. The siblings had rates similar to the general population. CONCLUSIONS: This pattern of findings suggests the presence of a gene in the WS region whose deletion predisposes to anxiety disorders. It is also worthwhile to investigate relations between genes deleted in WS and genes previously implicated in anxiety disorders. En ligne : http://dx.doi.org/10.1007/s11689-009-9003-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=341 [article] Anxiety disorders in children with williams syndrome, their mothers, and their siblings: implications for the etiology of anxiety disorders [Texte imprimé et/ou numérique] / O. LEYFER, Auteur ; J. WOODRUFF-BORDEN, Auteur ; C. B. MERVIS, Auteur . - p.4-14. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 1-1 (March 2009) . - p.4-14 Mots-clés : Anxiety  Family  Genetics  Intellectual disability  Mental retardation  Williams syndrome Index. décimale : PER Périodiques Résumé : PURPOSE: To determine the prevalence of anxiety disorders in children with Williams syndrome (WS), their sibling closest in age, and their mothers and to examine the predictors of anxiety in these groups. METHODS: The prevalence of anxiety disorders was assessed and compared to that in the general population. RESULTS: Children with WS had a significantly higher prevalence of specific phobia, generalized anxiety disorder (GAD), and separation anxiety in comparison to children in the general population. While mothers had a higher prevalence of GAD than population controls, the excess was accounted for by mothers who had onset after the birth of their WS child. The siblings had rates similar to the general population. CONCLUSIONS: This pattern of findings suggests the presence of a gene in the WS region whose deletion predisposes to anxiety disorders. It is also worthwhile to investigate relations between genes deleted in WS and genes previously implicated in anxiety disorders. En ligne : http://dx.doi.org/10.1007/s11689-009-9003-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=341

Autism Spectrum Symptomatology Among Children with Duplication 7q11.23 Syndrome / B. P. KLEIN-TASMAN in Journal of Autism and Developmental Disorders, 48-6 (June 2018)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 48-6 (June 2018) . - p.1982-1994 Titre : Autism Spectrum Symptomatology Among Children with Duplication 7q11.23 Syndrome Type de document : Texte imprimé et/ou numérique Auteurs : B. P. KLEIN-TASMAN, Auteur ; C. B. MERVIS, Auteur Article en page(s) : p.1982-1994 Langues : Anglais (eng) Mots-clés : 7q11.23 duplication syndrome  Autism spectrum disorders  Selective mutism  Social anxiety Index. décimale : PER Périodiques Résumé : Gold-standard diagnostic assessments of autism spectrum disorder (ASD) symptomatology were conducted on 63 children (mean CA: 8.81 years) with 7q11.23 duplication syndrome, one of the copy number variants identified by Sanders et al. (Neuron 70:863-885, 2011a) as associated with ASD. ASD classification rate was 39.6% for the Autism Diagnostic Interview-Revised and 25.4% for the Autism Diagnostic Observation Schedule-2 (ADOS-2). Based on these assessments combined with clinical judgment, 19.0% of children were diagnosed with ASD. Reasons for these discrepancies are discussed, as are differences in rate of diagnosis as a function of sex, age, and ADOS-2 module administered and differences in intellectual and adaptive behavior abilities as a function of presence or absence of ASD diagnosis and ADOS-2 module administered. Implications are addressed. En ligne : http://dx.doi.org/10.1007/s10803-017-3439-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=361 [article] Autism Spectrum Symptomatology Among Children with Duplication 7q11.23 Syndrome [Texte imprimé et/ou numérique] / B. P. KLEIN-TASMAN, Auteur ; C. B. MERVIS, Auteur . - p.1982-1994. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 48-6 (June 2018) . - p.1982-1994 Mots-clés : 7q11.23 duplication syndrome  Autism spectrum disorders  Selective mutism  Social anxiety Index. décimale : PER Périodiques Résumé : Gold-standard diagnostic assessments of autism spectrum disorder (ASD) symptomatology were conducted on 63 children (mean CA: 8.81 years) with 7q11.23 duplication syndrome, one of the copy number variants identified by Sanders et al. (Neuron 70:863-885, 2011a) as associated with ASD. ASD classification rate was 39.6% for the Autism Diagnostic Interview-Revised and 25.4% for the Autism Diagnostic Observation Schedule-2 (ADOS-2). Based on these assessments combined with clinical judgment, 19.0% of children were diagnosed with ASD. Reasons for these discrepancies are discussed, as are differences in rate of diagnosis as a function of sex, age, and ADOS-2 module administered and differences in intellectual and adaptive behavior abilities as a function of presence or absence of ASD diagnosis and ADOS-2 module administered. Implications are addressed. En ligne : http://dx.doi.org/10.1007/s10803-017-3439-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=361

Autism Spectrum Symptomatology in Children with Williams Syndrome Who Have Phrase Speech or Fluent Language / B. P. KLEIN-TASMAN in Journal of Autism and Developmental Disorders, 48-9 (September 2018)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 48-9 (September 2018) . - p.3037-3050 Titre : Autism Spectrum Symptomatology in Children with Williams Syndrome Who Have Phrase Speech or Fluent Language Type de document : Texte imprimé et/ou numérique Auteurs : B. P. KLEIN-TASMAN, Auteur ; F. VAN DER FLUIT, Auteur ; C. B. MERVIS, Auteur Article en page(s) : p.3037-3050 Langues : Anglais (eng) Mots-clés : Autism spectrum disorders  Intellectual disability  Social communication  Williams syndrome Index. décimale : PER Périodiques Résumé : To characterize autism spectrum-related symptomatology in children with Williams syndrome (WS) with phrase speech or fluent language, the Autism Diagnostic Observation Schedule Module 2 or 3 was administered. The cutoff for autism spectrum was met by 35% (14/40) who completed Module 2 and 30% (18/60) who completed Module 3. Similarities and differences in socio-communicative strengths and weaknesses as a function of language ability were identified. Symptom severity was negatively associated with IQ for participants with phrase speech but not for those with fluent language. The findings suggest an elevated risk of ASD for individuals with WS relative to the general population and contribute to a more nuanced sense of the socio-communicative functioning of children with WS. En ligne : http://dx.doi.org/10.1007/s10803-018-3555-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=367 [article] Autism Spectrum Symptomatology in Children with Williams Syndrome Who Have Phrase Speech or Fluent Language [Texte imprimé et/ou numérique] / B. P. KLEIN-TASMAN, Auteur ; F. VAN DER FLUIT, Auteur ; C. B. MERVIS, Auteur . - p.3037-3050. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 48-9 (September 2018) . - p.3037-3050 Mots-clés : Autism spectrum disorders  Intellectual disability  Social communication  Williams syndrome Index. décimale : PER Périodiques Résumé : To characterize autism spectrum-related symptomatology in children with Williams syndrome (WS) with phrase speech or fluent language, the Autism Diagnostic Observation Schedule Module 2 or 3 was administered. The cutoff for autism spectrum was met by 35% (14/40) who completed Module 2 and 30% (18/60) who completed Module 3. Similarities and differences in socio-communicative strengths and weaknesses as a function of language ability were identified. Symptom severity was negatively associated with IQ for participants with phrase speech but not for those with fluent language. The findings suggest an elevated risk of ASD for individuals with WS relative to the general population and contribute to a more nuanced sense of the socio-communicative functioning of children with WS. En ligne : http://dx.doi.org/10.1007/s10803-018-3555-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=367

The regulatory function of social referencing in preschoolers with Down syndrome or Williams syndrome / A. J. THURMAN in Journal of Neurodevelopmental Disorders, 5-1 (December 2013)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 5-1 (December 2013) . - p.2 Titre : The regulatory function of social referencing in preschoolers with Down syndrome or Williams syndrome Type de document : Texte imprimé et/ou numérique Auteurs : A. J. THURMAN, Auteur ; C. B. MERVIS, Auteur Article en page(s) : p.2 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : UNLABELLED: BACKGROUND: An important developmental task is to learn to recognize another person as a source of information and to utilize this information as a method of learning about the surrounding world. This socially guided form of learning, referred to as social referencing, is critical for the development of children's understanding of other people, themselves and their surrounding world. In the present project, the regulatory function of social referencing was examined in two genetic disorders that are characterized by differing patterns of socio-cognitive development: Down syndrome (DS) and Williams syndrome (WS). METHODS: Participants were 20 children with DS and 20 children with WS aged 42 to 71 months, matched on chronological age and gender. Each child participated in four studies: one study in which we examined performance in a social referencing paradigm and three studies in which we considered performance on tasks designed to tap each of three component abilities (initiating eye contact, gaze following and emotional responsivity) important for success in social referencing. RESULTS: The majority of children in both groups demonstrated positive behavioral responses regarding the stimulus in the Social Referencing task when the adult communicated a joyful message but did not regulate their own behavior in accordance with the adult's expression of fear. Between-group differences were observed in both conditions, with most differences indicating more advanced socio-communicative competence for children with DS than for children with WS even though the overall intellectual abilities and receptive language abilities of the children with WS were significantly higher than were those of the children with DS. The results of follow-up studies indicated that children with DS were more likely to initiate eye contact (unsolicited) and to follow another person's gaze in triadic situations than were children with WS. Neither group regulated their behavior in response to expressions of fear. CONCLUSIONS: These findings provide new insight into the development of the social cognitive phenotypes associated with DS and WS. These social cognitive differences found during the preschool years likely contribute to the differing phenotypes observed later in life between individuals with DS and individuals with WS. En ligne : http://dx.doi.org/10.1186/1866-1955-5-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=345 [article] The regulatory function of social referencing in preschoolers with Down syndrome or Williams syndrome [Texte imprimé et/ou numérique] / A. J. THURMAN, Auteur ; C. B. MERVIS, Auteur . - p.2. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 5-1 (December 2013) . - p.2 Index. décimale : PER Périodiques Résumé : UNLABELLED: BACKGROUND: An important developmental task is to learn to recognize another person as a source of information and to utilize this information as a method of learning about the surrounding world. This socially guided form of learning, referred to as social referencing, is critical for the development of children's understanding of other people, themselves and their surrounding world. In the present project, the regulatory function of social referencing was examined in two genetic disorders that are characterized by differing patterns of socio-cognitive development: Down syndrome (DS) and Williams syndrome (WS). METHODS: Participants were 20 children with DS and 20 children with WS aged 42 to 71 months, matched on chronological age and gender. Each child participated in four studies: one study in which we examined performance in a social referencing paradigm and three studies in which we considered performance on tasks designed to tap each of three component abilities (initiating eye contact, gaze following and emotional responsivity) important for success in social referencing. RESULTS: The majority of children in both groups demonstrated positive behavioral responses regarding the stimulus in the Social Referencing task when the adult communicated a joyful message but did not regulate their own behavior in accordance with the adult's expression of fear. Between-group differences were observed in both conditions, with most differences indicating more advanced socio-communicative competence for children with DS than for children with WS even though the overall intellectual abilities and receptive language abilities of the children with WS were significantly higher than were those of the children with DS. The results of follow-up studies indicated that children with DS were more likely to initiate eye contact (unsolicited) and to follow another person's gaze in triadic situations than were children with WS. Neither group regulated their behavior in response to expressions of fear. CONCLUSIONS: These findings provide new insight into the development of the social cognitive phenotypes associated with DS and WS. These social cognitive differences found during the preschool years likely contribute to the differing phenotypes observed later in life between individuals with DS and individuals with WS. En ligne : http://dx.doi.org/10.1186/1866-1955-5-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=345

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