ASD Comorbidity in Fragile X Syndrome: Symptom Profile and Predictors of Symptom Severity in Adolescent and Young Adult Males / Leonard ABBEDUTO in Journal of Autism and Developmental Disorders, 49-3 (March 2019)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 49-3 (March 2019) . - p.960-977 Titre : ASD Comorbidity in Fragile X Syndrome: Symptom Profile and Predictors of Symptom Severity in Adolescent and Young Adult Males Type de document : texte imprimé Auteurs : Leonard ABBEDUTO, Auteur ; Angela J. THURMAN, Auteur ; Andrea MCDUFFIE, Auteur ; Jessica KLUSEK, Auteur ; Robyn Tempero FEIGLES, Auteur ; W. Ted BROWN, Auteur ; Danielle J. HARVEY, Auteur ; Tatyana ADAYEV, Auteur ; Giuseppe LAFAUCI, Auteur ; Carl DOBKINS, Auteur ; Jane E. ROBERTS, Auteur Article en page(s) : p.960-977 Langues : Anglais (eng) Mots-clés : Autism spectrum disorder  Fmrp  Fragile X syndrome  Iq  Language  Psychiatric symptoms Index. décimale : PER Périodiques Résumé : Many males with FXS meet criteria for ASD. This study was designed to (1) describe ASD symptoms in adolescent and young adult males with FXS (n = 44) and (2) evaluate the contributions to ASD severity of cognitive, language, and psychiatric factors, as well as FMRP (the protein deficient in FXS). A few ASD symptoms on the ADOS-2 were universal in the sample. There was less impairment in restricted and repetitive behaviors (RRB) than in the social affective (SA) domain. The best predictor of overall ASD severity and SA severity was expressive syntactic ability. RRB severity was best predicted by the psychiatric factors. Implications for clinical practice and for understanding the ASD comorbidity in FXS are discussed. En ligne : http://dx.doi.org/10.1007/s10803-018-3796-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=386 [article] ASD Comorbidity in Fragile X Syndrome: Symptom Profile and Predictors of Symptom Severity in Adolescent and Young Adult Males [texte imprimé] / Leonard ABBEDUTO, Auteur ; Angela J. THURMAN, Auteur ; Andrea MCDUFFIE, Auteur ; Jessica KLUSEK, Auteur ; Robyn Tempero FEIGLES, Auteur ; W. Ted BROWN, Auteur ; Danielle J. HARVEY, Auteur ; Tatyana ADAYEV, Auteur ; Giuseppe LAFAUCI, Auteur ; Carl DOBKINS, Auteur ; Jane E. ROBERTS, Auteur . - p.960-977. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 49-3 (March 2019) . - p.960-977 Mots-clés : Autism spectrum disorder  Fmrp  Fragile X syndrome  Iq  Language  Psychiatric symptoms Index. décimale : PER Périodiques Résumé : Many males with FXS meet criteria for ASD. This study was designed to (1) describe ASD symptoms in adolescent and young adult males with FXS (n = 44) and (2) evaluate the contributions to ASD severity of cognitive, language, and psychiatric factors, as well as FMRP (the protein deficient in FXS). A few ASD symptoms on the ADOS-2 were universal in the sample. There was less impairment in restricted and repetitive behaviors (RRB) than in the social affective (SA) domain. The best predictor of overall ASD severity and SA severity was expressive syntactic ability. RRB severity was best predicted by the psychiatric factors. Implications for clinical practice and for understanding the ASD comorbidity in FXS are discussed. En ligne : http://dx.doi.org/10.1007/s10803-018-3796-2 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=386

Autism Symptomatology in Boys with Fragile X Syndrome: A Cross Sectional Developmental Trajectories Comparison with Nonsyndromic Autism Spectrum Disorder / Angela John THURMAN in Journal of Autism and Developmental Disorders, 45-9 (September 2015)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 45-9 (September 2015) . - p.2816-2832 Titre : Autism Symptomatology in Boys with Fragile X Syndrome: A Cross Sectional Developmental Trajectories Comparison with Nonsyndromic Autism Spectrum Disorder Type de document : texte imprimé Auteurs : Angela John THURMAN, Auteur ; Andrea MCDUFFIE, Auteur ; Sara T. KOVER, Auteur ; Randi J. HAGERMAN, Auteur ; Leonard ABBEDUTO, Auteur Article en page(s) : p.2816-2832 Langues : Anglais (eng) Mots-clés : Fragile X syndrome  Autism spectrum disorder  Behavioral phenotype  Nonverbal cognition  Expressive vocabulary  Developmental trajectory Index. décimale : PER Périodiques Résumé : Although males with fragile X syndrome (FXS) are frequently described as demonstrating autism symptomatology, there is much debate regarding whether the behavioral symptoms representing the core domains of autism are the result of the same or different underlying neurological/psychological mechanisms. The present study used a cross-sectional developmental trajectories approach to compare the profiles of autism symptomatology relative to chronological age (CA), nonverbal IQ, and expressive vocabulary ability between individuals with FXS and individuals with nonsyndromic ASD. Results suggest that the onset of autism symptoms and their developmental trajectories in males with FXS differ in important ways as a function of CA, nonverbal cognitive ability, and expressive vocabulary relative to males with nonsyndromic ASD. Theoretical and clinical implications are discussed. En ligne : http://dx.doi.org/10.1007/s10803-015-2443-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=267 [article] Autism Symptomatology in Boys with Fragile X Syndrome: A Cross Sectional Developmental Trajectories Comparison with Nonsyndromic Autism Spectrum Disorder [texte imprimé] / Angela John THURMAN, Auteur ; Andrea MCDUFFIE, Auteur ; Sara T. KOVER, Auteur ; Randi J. HAGERMAN, Auteur ; Leonard ABBEDUTO, Auteur . - p.2816-2832. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 45-9 (September 2015) . - p.2816-2832 Mots-clés : Fragile X syndrome  Autism spectrum disorder  Behavioral phenotype  Nonverbal cognition  Expressive vocabulary  Developmental trajectory Index. décimale : PER Périodiques Résumé : Although males with fragile X syndrome (FXS) are frequently described as demonstrating autism symptomatology, there is much debate regarding whether the behavioral symptoms representing the core domains of autism are the result of the same or different underlying neurological/psychological mechanisms. The present study used a cross-sectional developmental trajectories approach to compare the profiles of autism symptomatology relative to chronological age (CA), nonverbal IQ, and expressive vocabulary ability between individuals with FXS and individuals with nonsyndromic ASD. Results suggest that the onset of autism symptoms and their developmental trajectories in males with FXS differ in important ways as a function of CA, nonverbal cognitive ability, and expressive vocabulary relative to males with nonsyndromic ASD. Theoretical and clinical implications are discussed. En ligne : http://dx.doi.org/10.1007/s10803-015-2443-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=267

Brief Report: Parent Verbal Responsiveness and Language Development in Toddlers on the Autism Spectrum / Eileen HAEBIG in Journal of Autism and Developmental Disorders, 43-9 (September 2013)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 43-9 (September 2013) . - p.2218-2227 Titre : Brief Report: Parent Verbal Responsiveness and Language Development in Toddlers on the Autism Spectrum Type de document : texte imprimé Auteurs : Eileen HAEBIG, Auteur ; Andrea MCDUFFIE, Auteur ; Susan ELLIS WEISMER, Auteur Article en page(s) : p.2218-2227 Langues : Anglais (eng) Mots-clés : Autism  Parent responsiveness  Parent–child interactions Index. décimale : PER Périodiques Résumé : This study examined the longitudinal associations between parent verbal responsiveness and language 3 years later in 34 toddlers with a diagnosis of an autism spectrum disorder. Parent–child play samples were coded for child engagement and communication acts and for parent verbal responsiveness. Measures of responsive verbal behaviors were used to predict language gain scores 3 years later. Parent directives for language that followed into the child’s focus of attention were predictive of child receptive language gains. Parent comments that followed into the child’s focus of attention yielded differential effects depending on initial levels of child language. Children who were minimally verbal at age 2½ benefited from parent comments that followed into the their focus of attention, whereas children who were verbally fluent did not demonstrate such a benefit. En ligne : http://dx.doi.org/10.1007/s10803-013-1763-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=212 [article] Brief Report: Parent Verbal Responsiveness and Language Development in Toddlers on the Autism Spectrum [texte imprimé] / Eileen HAEBIG, Auteur ; Andrea MCDUFFIE, Auteur ; Susan ELLIS WEISMER, Auteur . - p.2218-2227. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 43-9 (September 2013) . - p.2218-2227 Mots-clés : Autism  Parent responsiveness  Parent–child interactions Index. décimale : PER Périodiques Résumé : This study examined the longitudinal associations between parent verbal responsiveness and language 3 years later in 34 toddlers with a diagnosis of an autism spectrum disorder. Parent–child play samples were coded for child engagement and communication acts and for parent verbal responsiveness. Measures of responsive verbal behaviors were used to predict language gain scores 3 years later. Parent directives for language that followed into the child’s focus of attention were predictive of child receptive language gains. Parent comments that followed into the child’s focus of attention yielded differential effects depending on initial levels of child language. Children who were minimally verbal at age 2½ benefited from parent comments that followed into the their focus of attention, whereas children who were verbally fluent did not demonstrate such a benefit. En ligne : http://dx.doi.org/10.1007/s10803-013-1763-5 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=212

A comparison of contexts for assessing joint attention in toddlers on the autism spectrum / Elizabeth M. ROOS in Autism, 12-3 (May 2008)  

Public ISBD [article]  inAutism > 12-3 (May 2008) . - p.275-291 Titre : A comparison of contexts for assessing joint attention in toddlers on the autism spectrum Type de document : texte imprimé Auteurs : Elizabeth M. ROOS, Auteur ; Andrea MCDUFFIE, Auteur ; Morton Ann GERNSBACHER, Auteur ; Susan ELLIS WEISMER, Auteur Année de publication : 2008 Article en page(s) : p.275-291 Langues : Anglais (eng) Mots-clés : autism joint-attention naturalistic-play social-communication Index. décimale : PER Périodiques Résumé : Children on the autism spectrum often demonstrate atypical joint attention, leading some researchers to consider joint attention defecits a core feature of the autism spectrum. Structured measures, such as the Early Social Communication Scales (ESCS), are commonly used to provide a metric of joint attention. To explore the assessment of joint attention in multiple contexts, we implemented an alternative system for coding joint attention behaviors. We compared initiation of joint attention (IJA) and response to joint attention (RJA) behaviors coded from naturalistic examiner—child play samples with similar IJA and RJA behaviors elicited within the structured ESCS protocol. Participants were 20 toddlers on the autism spectrum. Levels of IJA and RJA within the two assessment contexts were significantly and positively correlated, providing support for the use of naturalistic sampling of joint attention skills as a viable alternative, or supplement, to structured measures. En ligne : http://dx.doi.org/10.1177/1362361307089521 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=423 [article] A comparison of contexts for assessing joint attention in toddlers on the autism spectrum [texte imprimé] / Elizabeth M. ROOS, Auteur ; Andrea MCDUFFIE, Auteur ; Morton Ann GERNSBACHER, Auteur ; Susan ELLIS WEISMER, Auteur . - 2008 . - p.275-291. Langues : Anglais (eng) in Autism > 12-3 (May 2008) . - p.275-291 Mots-clés : autism joint-attention naturalistic-play social-communication Index. décimale : PER Périodiques Résumé : Children on the autism spectrum often demonstrate atypical joint attention, leading some researchers to consider joint attention defecits a core feature of the autism spectrum. Structured measures, such as the Early Social Communication Scales (ESCS), are commonly used to provide a metric of joint attention. To explore the assessment of joint attention in multiple contexts, we implemented an alternative system for coding joint attention behaviors. We compared initiation of joint attention (IJA) and response to joint attention (RJA) behaviors coded from naturalistic examiner—child play samples with similar IJA and RJA behaviors elicited within the structured ESCS protocol. Participants were 20 toddlers on the autism spectrum. Levels of IJA and RJA within the two assessment contexts were significantly and positively correlated, providing support for the use of naturalistic sampling of joint attention skills as a viable alternative, or supplement, to structured measures. En ligne : http://dx.doi.org/10.1177/1362361307089521 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=423

Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome / Angela John THURMAN in Journal of Neurodevelopmental Disorders, 12 (2020)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 12 (2020) Titre : Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome Type de document : texte imprimé Auteurs : Angela John THURMAN, Auteur ; Laura A. POTTER, Auteur ; Kyoungmi KIM, Auteur ; Flora TASSONE, Auteur ; Amy BANASIK, Auteur ; Sarah Nelson POTTER, Auteur ; Lauren BULLARD, Auteur ; Vivian NGUYEN, Auteur ; Andrea MCDUFFIE, Auteur ; Randi HAGERMAN, Auteur ; Leonard ABBEDUTO, Auteur Langues : Anglais (eng) Mots-clés : Adolescent  California  Child  Communication  Double-Blind Method  Female  Fragile X Syndrome/therapy  Humans  Language  Language Therapy/methods  Lovastatin/therapeutic use  Male  Mothers/education  Outcome Assessment, Health Care  Telecommunications  Distance teleconferencing  Expressive language sampling  Fragile X syndrome  Lovastatin  Narrative storytelling  Pili  Parent-implemented language intervention  outcome measures from Fulcrum Therapeutics and the Azrieli Foundation. FT has  received funding from the Azrieli Foundation, Zynerba, and Asuragen, Inc., for  studies in FXS. RH has received funding from Zynerba, Ovid, and the Azrieli  Foundation for treatment studies in children and adults with FXS. She has also  consulted with Zynerba and Fulcrum regarding treatment studies in FXS. LA has  received funding for the development and implementation of treatment outcome  measures from the F. Hoffmann-La Roche Ltd., Roche TCRC, Inc., Neuren  Pharmaceuticals Ltd., Fulcrum Therapeutics, Azrieli Foundation, and LuMind IDSC  Foundation. Index. décimale : PER Périodiques Résumé : BACKGROUND: The purpose of this study was to conduct a 20-week controlled trial of lovastatin (10 to 40 mg/day) in youth with fragile X syndrome (FXS) ages 10 to 17 years, combined with an open-label treatment of a parent-implemented language intervention (PILI), delivered via distance video teleconferencing to both treatment groups, lovastatin and placebo. METHOD: A randomized, double-blind trial was conducted at one site in the Sacramento, California, metropolitan area. Fourteen participants were assigned to the lovastatin group; two participants terminated early from the study. Sixteen participants were assigned to the placebo group. Lovastatin or placebo was administered orally in a capsule form, starting at 10 mg and increasing weekly or as tolerated by 10 mg increments, up to a maximum dose of 40 mg daily. A PILI was delivered to both groups for 12 weeks, with 4 activities per week, through video teleconferencing by an American Speech-Language Association-certified Speech-Language Pathologist, in collaboration with a Board-Certified Behavior Analyst. Parents were taught to use a set of language facilitation strategies while interacting with their children during a shared storytelling activity. The main outcome measures included absolute change from baseline to final visit in the means for youth total number of story-related utterances, youth number of different word roots, and parent total number of story-related utterances. RESULTS: Significant increases in all primary outcome measures were observed in both treatment groups. Significant improvements were also observed in parent reports of the severity of spoken language and social impairments in both treatment groups. In all cases, the amount of change observed did not differ across the two treatment groups. Although gains in parental use of the PILI-targeted intervention strategies were observed in both treatment groups, parental use of the PILI strategies was correlated with youth gains in the placebo group and not in the lovastatin group. CONCLUSION: Participants in both groups demonstrated significant changes in the primary outcome measures. The magnitude of change observed across the two groups was comparable, providing additional support for the efficacy of the use of PILI in youth with FXS. TRIAL REGISTRATION: US National Institutes of Health (ClinicalTrials.gov), NCT02642653. Registered 12/30/2015. En ligne : https://dx.doi.org/10.1186/s11689-020-09315-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573 [article] Controlled trial of lovastatin combined with an open-label treatment of a parent-implemented language intervention in youth with fragile X syndrome [texte imprimé] / Angela John THURMAN, Auteur ; Laura A. POTTER, Auteur ; Kyoungmi KIM, Auteur ; Flora TASSONE, Auteur ; Amy BANASIK, Auteur ; Sarah Nelson POTTER, Auteur ; Lauren BULLARD, Auteur ; Vivian NGUYEN, Auteur ; Andrea MCDUFFIE, Auteur ; Randi HAGERMAN, Auteur ; Leonard ABBEDUTO, Auteur. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 12 (2020) Mots-clés : Adolescent  California  Child  Communication  Double-Blind Method  Female  Fragile X Syndrome/therapy  Humans  Language  Language Therapy/methods  Lovastatin/therapeutic use  Male  Mothers/education  Outcome Assessment, Health Care  Telecommunications  Distance teleconferencing  Expressive language sampling  Fragile X syndrome  Lovastatin  Narrative storytelling  Pili  Parent-implemented language intervention  outcome measures from Fulcrum Therapeutics and the Azrieli Foundation. FT has  received funding from the Azrieli Foundation, Zynerba, and Asuragen, Inc., for  studies in FXS. RH has received funding from Zynerba, Ovid, and the Azrieli  Foundation for treatment studies in children and adults with FXS. She has also  consulted with Zynerba and Fulcrum regarding treatment studies in FXS. LA has  received funding for the development and implementation of treatment outcome  measures from the F. Hoffmann-La Roche Ltd., Roche TCRC, Inc., Neuren  Pharmaceuticals Ltd., Fulcrum Therapeutics, Azrieli Foundation, and LuMind IDSC  Foundation. Index. décimale : PER Périodiques Résumé : BACKGROUND: The purpose of this study was to conduct a 20-week controlled trial of lovastatin (10 to 40 mg/day) in youth with fragile X syndrome (FXS) ages 10 to 17 years, combined with an open-label treatment of a parent-implemented language intervention (PILI), delivered via distance video teleconferencing to both treatment groups, lovastatin and placebo. METHOD: A randomized, double-blind trial was conducted at one site in the Sacramento, California, metropolitan area. Fourteen participants were assigned to the lovastatin group; two participants terminated early from the study. Sixteen participants were assigned to the placebo group. Lovastatin or placebo was administered orally in a capsule form, starting at 10 mg and increasing weekly or as tolerated by 10 mg increments, up to a maximum dose of 40 mg daily. A PILI was delivered to both groups for 12 weeks, with 4 activities per week, through video teleconferencing by an American Speech-Language Association-certified Speech-Language Pathologist, in collaboration with a Board-Certified Behavior Analyst. Parents were taught to use a set of language facilitation strategies while interacting with their children during a shared storytelling activity. The main outcome measures included absolute change from baseline to final visit in the means for youth total number of story-related utterances, youth number of different word roots, and parent total number of story-related utterances. RESULTS: Significant increases in all primary outcome measures were observed in both treatment groups. Significant improvements were also observed in parent reports of the severity of spoken language and social impairments in both treatment groups. In all cases, the amount of change observed did not differ across the two treatment groups. Although gains in parental use of the PILI-targeted intervention strategies were observed in both treatment groups, parental use of the PILI strategies was correlated with youth gains in the placebo group and not in the lovastatin group. CONCLUSION: Participants in both groups demonstrated significant changes in the primary outcome measures. The magnitude of change observed across the two groups was comparable, providing additional support for the efficacy of the use of PILI in youth with FXS. TRIAL REGISTRATION: US National Institutes of Health (ClinicalTrials.gov), NCT02642653. Registered 12/30/2015. En ligne : https://dx.doi.org/10.1186/s11689-020-09315-4 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=573

Correction to: Expressive language sampling as a source of outcome measures for treatment studies in fragile X syndrome: feasibility, practice effects, test-retest reliability, and construct validity / Leonard ABBEDUTO in Journal of Neurodevelopmental Disorders, 12 (2020)  

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Developmental Correlates of Different Types of Motor Imitation in Young Children with Autism Spectrum Disorders / Andrea MCDUFFIE in Journal of Autism and Developmental Disorders, 37-3 (March 2007)  

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Distance delivery of a parent-implemented language intervention for young boys with fragile X syndrome / Lauren BULLARD in Autism & Developmental Language Impairments, 2 (January-December 2017)  

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Expressive language sampling as a source of outcome measures for treatment studies in fragile X syndrome: feasibility, practice effects, test-retest reliability, and construct validity / Leonard ABBEDUTO in Journal of Neurodevelopmental Disorders, 12 (2020)  

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Increasing verbal responsiveness in parents of children with autism:a pilot study / Courtney E. VENKER in Autism, 16-6 (November 2012)  

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Investigating Word Learning in Fragile X Syndrome: A Fast-Mapping Study / Andrea MCDUFFIE in Journal of Autism and Developmental Disorders, 43-7 (July 2013)  

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Language Skills of Males with Fragile X Syndrome or Nonsyndromic Autism Spectrum Disorder / Angela John THURMAN in Journal of Autism and Developmental Disorders, 47-3 (March 2017)  

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Learning Words in a Social World: Impairments Associated With ASD and Fragile X Syndrome / Andrea MCDUFFIE  

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Object interest in autism spectrum disorder: A treatment comparison / Andrea MCDUFFIE in Autism, 16-4 (July 2012)  

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Receptive Vocabulary in Boys with Autism Spectrum Disorder: Cross-Sectional Developmental Trajectories / Sara T. KOVER in Journal of Autism and Developmental Disorders, 43-11 (November 2013)  

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