Autism Spectrum Disorder Symptom Profiles in Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex and Neurofibromatosis Type 1 / Kyra LUBBERS in Journal of Autism and Developmental Disorders, 56-2 (February 2026)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 56-2 (February 2026) . - p.793-807 Titre : Autism Spectrum Disorder Symptom Profiles in Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex and Neurofibromatosis Type 1 Type de document : texte imprimé Auteurs : Kyra LUBBERS, Auteur ; Kamil R. HIRALAL, Auteur ; Gwendolyn C. DIELEMAN, Auteur ; Doesjka A. HAGENAAR, Auteur ; Bram DIERCKX, Auteur ; Jeroen S. LEGERSTEE, Auteur ; Pieter F. A. DE NIJS, Auteur ; André B. RIETMAN, Auteur ; Rianne OOSTENBRINK, Auteur ; Karen G. C. B. BINDELS-DE HEUS, Auteur ; Marie-Claire Y. DE WIT, Auteur ; Manon H. J. HILLEGERS, Auteur ; Leontine W. TEN HOOPEN, Auteur ; Sabine E. MOUS, Auteur Article en page(s) : p.793-807 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Studying Autism Spectrum Disorder (ASD) heterogeneity in biologically homogeneous samples may increase our knowledge of ASD etiology. Fragile X syndrome (FXS), Angelman syndrome (AS), Tuberous Sclerosis Complex (TSC), and Neurofibromatosis type 1 (NF1) are monogenic disorders with high a prevalence of ASD symptomatology. This study aimed to identify ASD symptom profiles in a large group of children and adolescents (0;9–28 years) with FXS, AS, TSC, and NF1. Data on ASD symptomatology (Autism Diagnostic Observation Scale (ADOS-2) & Social Responsiveness Scale (SRS-2)) were collected from children and adolescents with FXS (n = 54), AS (n = 93), TSC (n = 112), and NF1 (n = 278). To identify groups of individuals with similar ASD profiles, we performed two latent profile analyses. We identified a four-profile model based on the ADOS-2, with a (1) ‘Non-spectrum symptom profile’, (2) ‘Social Affect symptom profile’, (3)‘Restricted/Repetitive Behaviors symptom profile’, and (4)‘ASD symptom profile’. We also identified a four-profile model based on the SRS, with a (1)‘Non-clinical symptom profile’, (2)‘Mild symptom profile’, (3)‘Moderate symptom profile’, and (4)‘Severe symptom profile’. Although each syndrome group exhibited varying degrees of severity, they also displayed heterogeneity in the profiles in which they were classified. We found distinct ASD symptom profiles in a population consisting of children and adolescents with FXS, AS, TSC, and NF1. Our study highlights the importance of a personalized approach to the identification and management of ASD symptoms in rare genetic syndromes. Future studies should aim to include more domains of functioning and investigate the stability of latent profiles over time. En ligne : https://doi.org/10.1007/s10803-024-06557-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=580 [article] Autism Spectrum Disorder Symptom Profiles in Fragile X Syndrome, Angelman Syndrome, Tuberous Sclerosis Complex and Neurofibromatosis Type 1 [texte imprimé] / Kyra LUBBERS, Auteur ; Kamil R. HIRALAL, Auteur ; Gwendolyn C. DIELEMAN, Auteur ; Doesjka A. HAGENAAR, Auteur ; Bram DIERCKX, Auteur ; Jeroen S. LEGERSTEE, Auteur ; Pieter F. A. DE NIJS, Auteur ; André B. RIETMAN, Auteur ; Rianne OOSTENBRINK, Auteur ; Karen G. C. B. BINDELS-DE HEUS, Auteur ; Marie-Claire Y. DE WIT, Auteur ; Manon H. J. HILLEGERS, Auteur ; Leontine W. TEN HOOPEN, Auteur ; Sabine E. MOUS, Auteur . - p.793-807. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 56-2 (February 2026) . - p.793-807 Index. décimale : PER Périodiques Résumé : Studying Autism Spectrum Disorder (ASD) heterogeneity in biologically homogeneous samples may increase our knowledge of ASD etiology. Fragile X syndrome (FXS), Angelman syndrome (AS), Tuberous Sclerosis Complex (TSC), and Neurofibromatosis type 1 (NF1) are monogenic disorders with high a prevalence of ASD symptomatology. This study aimed to identify ASD symptom profiles in a large group of children and adolescents (0;9–28 years) with FXS, AS, TSC, and NF1. Data on ASD symptomatology (Autism Diagnostic Observation Scale (ADOS-2) & Social Responsiveness Scale (SRS-2)) were collected from children and adolescents with FXS (n = 54), AS (n = 93), TSC (n = 112), and NF1 (n = 278). To identify groups of individuals with similar ASD profiles, we performed two latent profile analyses. We identified a four-profile model based on the ADOS-2, with a (1) ‘Non-spectrum symptom profile’, (2) ‘Social Affect symptom profile’, (3)‘Restricted/Repetitive Behaviors symptom profile’, and (4)‘ASD symptom profile’. We also identified a four-profile model based on the SRS, with a (1)‘Non-clinical symptom profile’, (2)‘Mild symptom profile’, (3)‘Moderate symptom profile’, and (4)‘Severe symptom profile’. Although each syndrome group exhibited varying degrees of severity, they also displayed heterogeneity in the profiles in which they were classified. We found distinct ASD symptom profiles in a population consisting of children and adolescents with FXS, AS, TSC, and NF1. Our study highlights the importance of a personalized approach to the identification and management of ASD symptoms in rare genetic syndromes. Future studies should aim to include more domains of functioning and investigate the stability of latent profiles over time. En ligne : https://doi.org/10.1007/s10803-024-06557-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=580

CBT for childhood anxiety disorders: differential changes in selective attention between treatment responders and non-responders / Jeroen S. LEGERSTEE in Journal of Child Psychology and Psychiatry, 51-2 (February 2010)  

Public ISBD [article]  inJournal of Child Psychology and Psychiatry > 51-2 (February 2010) . - p.162-172 Titre : CBT for childhood anxiety disorders: differential changes in selective attention between treatment responders and non-responders Type de document : texte imprimé Auteurs : Jeroen S. LEGERSTEE, Auteur ; Frank C. VERHULST, Auteur ; Philip D.A. TREFFERS, Auteur ; Elisabeth M.W.J. UTENS, Auteur ; Joke H.M. TULEN, Auteur ; Bram DIERCKX, Auteur Année de publication : 2010 Article en page(s) : p.162-172 Langues : Anglais (eng) Mots-clés : Childhood-anxiety-disorders selective-attention cognitive-behavioural-therapy dot-probe-task Index. décimale : PER Périodiques Résumé : Background: This study examined whether treatment response to stepped-care cognitive-behavioural treatment (CBT) is associated with changes in threat-related selective attention and its specific components in a large clinical sample of anxiety-disordered children. Methods: Ninety-one children with an anxiety disorder were included in the present study. Children received a standardized stepped-care CBT. Three treatment response groups were distinguished: initial responders (anxiety disorder free after phase one: child-focused CBT), secondary responders (anxiety disorder free after phase two: child–parent-focused CBT), and treatment non-responders. Treatment response was determined using a semi-structured clinical interview. Children performed a pictorial dot-probe task before and after stepped-care CBT (i.e., before phase one and after phase two CBT). Results: Changes in selective attention to severely threatening pictures, but not to mildly threatening pictures, were significantly associated with treatment success. At pre-treatment assessment, initial responders selectively attended away from severely threatening pictures, whereas secondary responders selectively attended toward severely threatening pictures. After stepped-care CBT, initial and secondary responders did not show any selectivity in the attentional processing of severely threatening pictures. Treatment non-responders did not show any changes in selective attention due to CBT. Conclusions: Initial and secondary treatment responders showed a reduction of their predisposition to selectively attend away or toward severely threatening pictures, respectively. Treatment non-responders did not show any changes in selective attention. The pictorial dot-probe task can be considered a potentially valuable tool in assigning children to appropriate treatment formats as well as for monitoring changes in selective attention during the course of CBT. En ligne : http://dx.doi.org/10.1111/j.1469-7610.2009.02143.x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=941 [article] CBT for childhood anxiety disorders: differential changes in selective attention between treatment responders and non-responders [texte imprimé] / Jeroen S. LEGERSTEE, Auteur ; Frank C. VERHULST, Auteur ; Philip D.A. TREFFERS, Auteur ; Elisabeth M.W.J. UTENS, Auteur ; Joke H.M. TULEN, Auteur ; Bram DIERCKX, Auteur . - 2010 . - p.162-172. Langues : Anglais (eng) in Journal of Child Psychology and Psychiatry > 51-2 (February 2010) . - p.162-172 Mots-clés : Childhood-anxiety-disorders selective-attention cognitive-behavioural-therapy dot-probe-task Index. décimale : PER Périodiques Résumé : Background: This study examined whether treatment response to stepped-care cognitive-behavioural treatment (CBT) is associated with changes in threat-related selective attention and its specific components in a large clinical sample of anxiety-disordered children. Methods: Ninety-one children with an anxiety disorder were included in the present study. Children received a standardized stepped-care CBT. Three treatment response groups were distinguished: initial responders (anxiety disorder free after phase one: child-focused CBT), secondary responders (anxiety disorder free after phase two: child–parent-focused CBT), and treatment non-responders. Treatment response was determined using a semi-structured clinical interview. Children performed a pictorial dot-probe task before and after stepped-care CBT (i.e., before phase one and after phase two CBT). Results: Changes in selective attention to severely threatening pictures, but not to mildly threatening pictures, were significantly associated with treatment success. At pre-treatment assessment, initial responders selectively attended away from severely threatening pictures, whereas secondary responders selectively attended toward severely threatening pictures. After stepped-care CBT, initial and secondary responders did not show any selectivity in the attentional processing of severely threatening pictures. Treatment non-responders did not show any changes in selective attention due to CBT. Conclusions: Initial and secondary treatment responders showed a reduction of their predisposition to selectively attend away or toward severely threatening pictures, respectively. Treatment non-responders did not show any changes in selective attention. The pictorial dot-probe task can be considered a potentially valuable tool in assigning children to appropriate treatment formats as well as for monitoring changes in selective attention during the course of CBT. En ligne : http://dx.doi.org/10.1111/j.1469-7610.2009.02143.x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=941

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