Autism Symptoms Across Adulthood in Men with Fragile X Syndrome: A Cross-Sectional Analysis / Sigan L. HARTLEY in Journal of Autism and Developmental Disorders, 45-11 (November 2015)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 45-11 (November 2015) . - p.3668-3679 Titre : Autism Symptoms Across Adulthood in Men with Fragile X Syndrome: A Cross-Sectional Analysis Type de document : Texte imprimé et/ou numérique Auteurs : Sigan L. HARTLEY, Auteur ; Anne C. WHEELER, Auteur ; Marsha R. MAILICK, Auteur ; Melissa RASPA, Auteur ; Iulia MIHAILA, Auteur ; Ellen BISHOP, Auteur ; Donald B. Jr BAILEY, Auteur Article en page(s) : p.3668-3679 Langues : Anglais (eng) Mots-clés : Fragile X syndrome  Autism  DSM  Adult  Aging Index. décimale : PER Périodiques Résumé : A cross-sectional analysis was used to examine age-related differences in ASD symptoms and corresponding differences in disruptive behavior and social skills in 281 adult men with fragile X syndrome. Four age groups were created: 18–21, 22–29, 30–39, and 40–49 years. The 18–21 year-old group was reported to have more impairments in verbal communication than the 22–29 year-old group and more restricted and repetitive behaviors than the 40–49 year-old group. There was not an age-group difference in the percentage of men who met criteria for an ASD diagnosis based on respondent-reported, current symptoms. There was a trend for an age-related difference in disruptive behavior. Findings add to understanding of the developmental trajectory of ASD symptoms in adulthood. En ligne : http://dx.doi.org/10.1007/s10803-015-2513-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=270 [article] Autism Symptoms Across Adulthood in Men with Fragile X Syndrome: A Cross-Sectional Analysis [Texte imprimé et/ou numérique] / Sigan L. HARTLEY, Auteur ; Anne C. WHEELER, Auteur ; Marsha R. MAILICK, Auteur ; Melissa RASPA, Auteur ; Iulia MIHAILA, Auteur ; Ellen BISHOP, Auteur ; Donald B. Jr BAILEY, Auteur . - p.3668-3679. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 45-11 (November 2015) . - p.3668-3679 Mots-clés : Fragile X syndrome  Autism  DSM  Adult  Aging Index. décimale : PER Périodiques Résumé : A cross-sectional analysis was used to examine age-related differences in ASD symptoms and corresponding differences in disruptive behavior and social skills in 281 adult men with fragile X syndrome. Four age groups were created: 18–21, 22–29, 30–39, and 40–49 years. The 18–21 year-old group was reported to have more impairments in verbal communication than the 22–29 year-old group and more restricted and repetitive behaviors than the 40–49 year-old group. There was not an age-group difference in the percentage of men who met criteria for an ASD diagnosis based on respondent-reported, current symptoms. There was a trend for an age-related difference in disruptive behavior. Findings add to understanding of the developmental trajectory of ASD symptoms in adulthood. En ligne : http://dx.doi.org/10.1007/s10803-015-2513-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=270

Decisional Capacity for Informed Consent in Males and Females with Fragile X Syndrome / Anne C. WHEELER in Journal of Autism and Developmental Disorders, 50-5 (May 2020)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 50-5 (May 2020) . - p.1725-1747 Titre : Decisional Capacity for Informed Consent in Males and Females with Fragile X Syndrome Type de document : Texte imprimé et/ou numérique Auteurs : Anne C. WHEELER, Auteur ; Amanda WYLIE, Auteur ; Melissa RASPA, Auteur ; Adrienne VILLAGOMEZ, Auteur ; Kylee MILLER, Auteur ; Anne EDWARDS, Auteur ; Margaret L. DERAMUS, Auteur ; Paul S. APPELBAUM, Auteur ; Donald B. Jr BAILEY, Auteur Article en page(s) : p.1725-1747 Langues : Anglais (eng) Mots-clés : Clinical trials  Decisional capacity  Fragile X syndrome  Informed consent Index. décimale : PER Périodiques Résumé : Although informed consent is critical for all research, there is increased ethical responsibility as individuals with intellectual or developmental disabilities (IDD) become the focus of more clinical trials. This study examined decisional capacity for informed consent to clinical trials in individuals with fragile X syndrome (FXS). Participants were 152 adolescents and adults (80 males, 72 females) with FXS who completed a measure of decisional capacity and a comprehensive battery of neurocognitive and psychiatric measures. Females outperformed males on all aspects of decisional capacity. The ability to understand aspects of the clinical trial had the strongest association with the ability to appreciate and reason about the decision. Scaffolding improved understanding, suggesting researchers can take steps to improve decisional capacity and the informed consent process. En ligne : http://dx.doi.org/10.1007/s10803-019-03930-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=422 [article] Decisional Capacity for Informed Consent in Males and Females with Fragile X Syndrome [Texte imprimé et/ou numérique] / Anne C. WHEELER, Auteur ; Amanda WYLIE, Auteur ; Melissa RASPA, Auteur ; Adrienne VILLAGOMEZ, Auteur ; Kylee MILLER, Auteur ; Anne EDWARDS, Auteur ; Margaret L. DERAMUS, Auteur ; Paul S. APPELBAUM, Auteur ; Donald B. Jr BAILEY, Auteur . - p.1725-1747. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 50-5 (May 2020) . - p.1725-1747 Mots-clés : Clinical trials  Decisional capacity  Fragile X syndrome  Informed consent Index. décimale : PER Périodiques Résumé : Although informed consent is critical for all research, there is increased ethical responsibility as individuals with intellectual or developmental disabilities (IDD) become the focus of more clinical trials. This study examined decisional capacity for informed consent to clinical trials in individuals with fragile X syndrome (FXS). Participants were 152 adolescents and adults (80 males, 72 females) with FXS who completed a measure of decisional capacity and a comprehensive battery of neurocognitive and psychiatric measures. Females outperformed males on all aspects of decisional capacity. The ability to understand aspects of the clinical trial had the strongest association with the ability to appreciate and reason about the decision. Scaffolding improved understanding, suggesting researchers can take steps to improve decisional capacity and the informed consent process. En ligne : http://dx.doi.org/10.1007/s10803-019-03930-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=422

DSM-5 Changes and the Prevalence of Parent-Reported Autism Spectrum Symptoms in Fragile X Syndrome / Anne C. WHEELER in Journal of Autism and Developmental Disorders, 45-3 (March 2015)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 45-3 (March 2015) . - p.816-829 Titre : DSM-5 Changes and the Prevalence of Parent-Reported Autism Spectrum Symptoms in Fragile X Syndrome Type de document : Texte imprimé et/ou numérique Auteurs : Anne C. WHEELER, Auteur ; Joanna MUSSEY, Auteur ; Adrienne VILLAGOMEZ, Auteur ; Ellen BISHOP, Auteur ; Melissa RASPA, Auteur ; Anne EDWARDS, Auteur ; James W. BODFISH, Auteur ; Carla M. BANN, Auteur ; Donald B. Jr BAILEY, Auteur Article en page(s) : p.816-829 Langues : Anglais (eng) Mots-clés : Fragile X syndrome  DSM-5 criteria  Autism spectrum disorder diagnoses Index. décimale : PER Périodiques Résumé : We used survey methodology to assess parent-reported autism symptomology in 758 individuals (639 males; 119 females) with fragile X syndrome (FXS). Caregivers reported whether their child with FXS had been diagnosed with an autism spectrum disorder (ASD) and endorsed symptoms based on a list of observable behaviors related to ASD diagnoses. Symptom counts were categorized based on DSM-IV-TR and DSM-5 criteria. Based on behavioral symptoms endorsed by caregivers, 38.7 % of males and 24.7 % of females met criteria for DSM-IV-TR diagnosis of autistic disorder. Significantly fewer males (27.8 %) and females (11.3 %) met criteria for ASD based on DSM-5 criteria. Although 86.4 % of males and 61.7 % of females met criteria for the restricted and repetitive behavior domain for DSM-5, only 29.4 % of males and 13.0 % of females met criteria for the social communication and interaction (SCI) domain. Relaxing the social communication criteria by one symptom count led to a threefold increase in those meeting criteria for ASD, suggesting the importance of subthreshold SCI symptoms for individuals with FXS in ASD diagnoses. Findings suggest important differences in the way ASD may be conceptualized in FXS based on the new DSM-5 criteria. En ligne : http://dx.doi.org/10.1007/s10803-014-2246-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=258 [article] DSM-5 Changes and the Prevalence of Parent-Reported Autism Spectrum Symptoms in Fragile X Syndrome [Texte imprimé et/ou numérique] / Anne C. WHEELER, Auteur ; Joanna MUSSEY, Auteur ; Adrienne VILLAGOMEZ, Auteur ; Ellen BISHOP, Auteur ; Melissa RASPA, Auteur ; Anne EDWARDS, Auteur ; James W. BODFISH, Auteur ; Carla M. BANN, Auteur ; Donald B. Jr BAILEY, Auteur . - p.816-829. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 45-3 (March 2015) . - p.816-829 Mots-clés : Fragile X syndrome  DSM-5 criteria  Autism spectrum disorder diagnoses Index. décimale : PER Périodiques Résumé : We used survey methodology to assess parent-reported autism symptomology in 758 individuals (639 males; 119 females) with fragile X syndrome (FXS). Caregivers reported whether their child with FXS had been diagnosed with an autism spectrum disorder (ASD) and endorsed symptoms based on a list of observable behaviors related to ASD diagnoses. Symptom counts were categorized based on DSM-IV-TR and DSM-5 criteria. Based on behavioral symptoms endorsed by caregivers, 38.7 % of males and 24.7 % of females met criteria for DSM-IV-TR diagnosis of autistic disorder. Significantly fewer males (27.8 %) and females (11.3 %) met criteria for ASD based on DSM-5 criteria. Although 86.4 % of males and 61.7 % of females met criteria for the restricted and repetitive behavior domain for DSM-5, only 29.4 % of males and 13.0 % of females met criteria for the social communication and interaction (SCI) domain. Relaxing the social communication criteria by one symptom count led to a threefold increase in those meeting criteria for ASD, suggesting the importance of subthreshold SCI symptoms for individuals with FXS in ASD diagnoses. Findings suggest important differences in the way ASD may be conceptualized in FXS based on the new DSM-5 criteria. En ligne : http://dx.doi.org/10.1007/s10803-014-2246-z Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=258

Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome / Melissa RASPA ; Carla M. BANN ; Julia M. GABLE ; Holly K. HARRIS ; Dejan B. BUDIMIROVIC ; Reymundo LOZANO ; Elizabeth BERRY-KRAVIS ; Milen VELINOV ; Amy L. TALBOY ; Stephanie L. SHERMAN ; Walter E. KAUFMANN ; Marcy SCHUSTER ; Nicole TARTAGLIA ; Robyn A. FILIPINK ; Dejan B. BUDIMIROVIC ; Deborah BARBOUTH ; Amy LIGHTBODY ; Allan REISS ; Carol M. DELAHUNTY ; Randi J. HAGERMAN ; David HESSL ; Craig A. ERICKSON ; Gary FELDMAN ; Jonathan D. PICKER ; Ave M. LACHIEWICZ ; Holly K. HARRIS ; Amy ESLER ; Richard E. FRYE ; Patricia A. EVANS ; Mary Ann MORRIS ; Barbara A. HAAS-GIVLER ; Andrea L. GROPMAN ; Ryan S. UY ; Carrie BUCHANAN ; Jean A. FRAZIER ; Stephanie M. MORRIS ; Forward CONSORTIUM in Journal of Autism and Developmental Disorders, 54-2 (February 2024)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 54-2 (February 2024) . - p.725-737 Titre : Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome Type de document : Texte imprimé et/ou numérique Auteurs : Melissa RASPA, Auteur ; Carla M. BANN, Auteur ; Julia M. GABLE, Auteur ; Holly K. HARRIS, Auteur ; Dejan B. BUDIMIROVIC, Auteur ; Reymundo LOZANO, Auteur ; Elizabeth BERRY-KRAVIS, Auteur ; Milen VELINOV, Auteur ; Amy L. TALBOY, Auteur ; Stephanie L. SHERMAN, Auteur ; Walter E. KAUFMANN, Auteur ; Marcy SCHUSTER, Auteur ; Nicole TARTAGLIA, Auteur ; Robyn A. FILIPINK, Auteur ; Dejan B. BUDIMIROVIC, Auteur ; Deborah BARBOUTH, Auteur ; Amy LIGHTBODY, Auteur ; Allan REISS, Auteur ; Carol M. DELAHUNTY, Auteur ; Randi J. HAGERMAN, Auteur ; David HESSL, Auteur ; Craig A. ERICKSON, Auteur ; Gary FELDMAN, Auteur ; Jonathan D. PICKER, Auteur ; Ave M. LACHIEWICZ, Auteur ; Holly K. HARRIS, Auteur ; Amy ESLER, Auteur ; Richard E. FRYE, Auteur ; Patricia A. EVANS, Auteur ; Mary Ann MORRIS, Auteur ; Barbara A. HAAS-GIVLER, Auteur ; Andrea L. GROPMAN, Auteur ; Ryan S. UY, Auteur ; Carrie BUCHANAN, Auteur ; Jean A. FRAZIER, Auteur ; Stephanie M. MORRIS, Auteur ; Forward CONSORTIUM, Auteur Article en page(s) : p.725-737 Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is characterized by variable neurobehavioral abnormalities, which leads to difficulties in developing and evaluating treatments and in determining accurate prognosis. We employed a pediatric cross-sectional sample (1,072 males, 338 females) from FORWARD, a clinic-based natural history study, to identify behavioral subtypes by latent class analysis. Input included co-occurring behavioral conditions, sleep and sensory problems, autistic behavior scales (SCQ, SRS-2), and the Aberrant Behavior Checklist revised for FXS (ABCFX). A 5-class solution yielded the most clinically meaningful, pharmacotherapy independent behavioral groups with distinctive SCQ, SRS-2, and ABCFX profiles, and adequate non-overlap (??71%): ?Mild? (31%), ?Moderate without Social Impairment? (32%), ?Moderate with Social Impairment? (7%), ?Moderate with Disruptive Behavior? (20%), and ?Severe? (9%). Our findings support FXS subtyping, for improving clinical management and therapeutic development. En ligne : https://doi.org/10.1007/s10803-022-05821-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=520 [article] Latent Class Analysis Identifies Distinctive Behavioral Subtypes in Children with Fragile X Syndrome [Texte imprimé et/ou numérique] / Melissa RASPA, Auteur ; Carla M. BANN, Auteur ; Julia M. GABLE, Auteur ; Holly K. HARRIS, Auteur ; Dejan B. BUDIMIROVIC, Auteur ; Reymundo LOZANO, Auteur ; Elizabeth BERRY-KRAVIS, Auteur ; Milen VELINOV, Auteur ; Amy L. TALBOY, Auteur ; Stephanie L. SHERMAN, Auteur ; Walter E. KAUFMANN, Auteur ; Marcy SCHUSTER, Auteur ; Nicole TARTAGLIA, Auteur ; Robyn A. FILIPINK, Auteur ; Dejan B. BUDIMIROVIC, Auteur ; Deborah BARBOUTH, Auteur ; Amy LIGHTBODY, Auteur ; Allan REISS, Auteur ; Carol M. DELAHUNTY, Auteur ; Randi J. HAGERMAN, Auteur ; David HESSL, Auteur ; Craig A. ERICKSON, Auteur ; Gary FELDMAN, Auteur ; Jonathan D. PICKER, Auteur ; Ave M. LACHIEWICZ, Auteur ; Holly K. HARRIS, Auteur ; Amy ESLER, Auteur ; Richard E. FRYE, Auteur ; Patricia A. EVANS, Auteur ; Mary Ann MORRIS, Auteur ; Barbara A. HAAS-GIVLER, Auteur ; Andrea L. GROPMAN, Auteur ; Ryan S. UY, Auteur ; Carrie BUCHANAN, Auteur ; Jean A. FRAZIER, Auteur ; Stephanie M. MORRIS, Auteur ; Forward CONSORTIUM, Auteur . - p.725-737. in Journal of Autism and Developmental Disorders > 54-2 (February 2024) . - p.725-737 Index. décimale : PER Périodiques Résumé : Fragile X syndrome (FXS) is characterized by variable neurobehavioral abnormalities, which leads to difficulties in developing and evaluating treatments and in determining accurate prognosis. We employed a pediatric cross-sectional sample (1,072 males, 338 females) from FORWARD, a clinic-based natural history study, to identify behavioral subtypes by latent class analysis. Input included co-occurring behavioral conditions, sleep and sensory problems, autistic behavior scales (SCQ, SRS-2), and the Aberrant Behavior Checklist revised for FXS (ABCFX). A 5-class solution yielded the most clinically meaningful, pharmacotherapy independent behavioral groups with distinctive SCQ, SRS-2, and ABCFX profiles, and adequate non-overlap (??71%): ?Mild? (31%), ?Moderate without Social Impairment? (32%), ?Moderate with Social Impairment? (7%), ?Moderate with Disruptive Behavior? (20%), and ?Severe? (9%). Our findings support FXS subtyping, for improving clinical management and therapeutic development. En ligne : https://doi.org/10.1007/s10803-022-05821-7 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=520

Psychometric Assessment of the Rett Syndrome Caregiver Assessment of Symptom Severity (RCASS) / Melissa RASPA in Journal of Autism and Developmental Disorders, 55-3 (March 2025)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 55-3 (March 2025) . - p.997-1009 Titre : Psychometric Assessment of the Rett Syndrome Caregiver Assessment of Symptom Severity (RCASS) Type de document : Texte imprimé et/ou numérique Auteurs : Melissa RASPA, Auteur ; Angela GWALTNEY, Auteur ; Carla BANN, Auteur ; Jana VON HEHN, Auteur ; Timothy A. BENKE, Auteur ; Eric D. MARSH, Auteur ; Sarika U. PETERS, Auteur ; Amitha ANANTH, Auteur ; Alan K. PERCY, Auteur ; Jeffrey L. NEUL, Auteur Article en page(s) : p.997-1009 Langues : Anglais (eng) Index. décimale : PER Périodiques Résumé : Rett syndrome is a severe neurodevelopmental disorder that affects about 1 in 10,000 females. Clinical trials of disease modifying therapies are on the rise, but there are few psychometrically sound caregiver-reported outcome measures available to assess treatment benefit. We report on a new caregiver-reported outcome measure, the Rett Caregiver Assessment of Symptom Severity (RCASS). Using data from the Rett Natural History Study (n = 649), we examined the factor structure, using both exploratory and confirmatory factor analysis, and the reliability and validity of the RCASS. The four-factor model had the best overall fit, which covered movement, communication, behavior, and Rett-specific symptoms. The RCASS had moderate internal consistency. Strong face validity was found with age and mutation type, and convergent validity was established with other similar measures, including the Revised Motor-Behavior Assessment Scale, Clinical Severity Scale, Clinical Global Impression Scale, and the Child Health Questionnaire. These data provide initial evidence that the RCASS is a viable caregiver-outcome measure for use in clinical trials in Rett syndrome. Future work to assess sensitivity to change and other measures of reliability, such as test-retest and inter-rater agreement, are needed. En ligne : https://doi.org/10.1007/s10803-024-06238-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=548 [article] Psychometric Assessment of the Rett Syndrome Caregiver Assessment of Symptom Severity (RCASS) [Texte imprimé et/ou numérique] / Melissa RASPA, Auteur ; Angela GWALTNEY, Auteur ; Carla BANN, Auteur ; Jana VON HEHN, Auteur ; Timothy A. BENKE, Auteur ; Eric D. MARSH, Auteur ; Sarika U. PETERS, Auteur ; Amitha ANANTH, Auteur ; Alan K. PERCY, Auteur ; Jeffrey L. NEUL, Auteur . - p.997-1009. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 55-3 (March 2025) . - p.997-1009 Index. décimale : PER Périodiques Résumé : Rett syndrome is a severe neurodevelopmental disorder that affects about 1 in 10,000 females. Clinical trials of disease modifying therapies are on the rise, but there are few psychometrically sound caregiver-reported outcome measures available to assess treatment benefit. We report on a new caregiver-reported outcome measure, the Rett Caregiver Assessment of Symptom Severity (RCASS). Using data from the Rett Natural History Study (n = 649), we examined the factor structure, using both exploratory and confirmatory factor analysis, and the reliability and validity of the RCASS. The four-factor model had the best overall fit, which covered movement, communication, behavior, and Rett-specific symptoms. The RCASS had moderate internal consistency. Strong face validity was found with age and mutation type, and convergent validity was established with other similar measures, including the Revised Motor-Behavior Assessment Scale, Clinical Severity Scale, Clinical Global Impression Scale, and the Child Health Questionnaire. These data provide initial evidence that the RCASS is a viable caregiver-outcome measure for use in clinical trials in Rett syndrome. Future work to assess sensitivity to change and other measures of reliability, such as test-retest and inter-rater agreement, are needed. En ligne : https://doi.org/10.1007/s10803-024-06238-0 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=548

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