Brief Report: Social Disability in Autism Spectrum Disorder: Results from Research Units on Pediatric Psychopharmacology (RUPP) Autism Network Trials / Lawrence SCAHILL in Journal of Autism and Developmental Disorders, 43-3 (March 2013)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 43-3 (March 2013) . - p.739-746 Titre : Brief Report: Social Disability in Autism Spectrum Disorder: Results from Research Units on Pediatric Psychopharmacology (RUPP) Autism Network Trials Type de document : texte imprimé Auteurs : Lawrence SCAHILL, Auteur ; Victoria HALLETT, Auteur ; Michael G. AMAN, Auteur ; Christopher J. MCDOUGLE, Auteur ; L. Eugene ARNOLD, Auteur ; James T. MCCRACKEN, Auteur ; Elaine TIERNEY, Auteur ; Yanhong DENG, Auteur ; James DZIURA, Auteur ; Benedetto VITIELLO, Auteur Article en page(s) : p.739-746 Langues : Anglais (eng) Mots-clés : Social disability  Autism  Research Units on Pediatric Psychopharmacology (RUPP)  Autism Network  Social withdrawal  Risperidone  Aberrant Behavior Checklist Index. décimale : PER Périodiques Résumé : There is growing interest in measuring social disability as a core element of autism spectrum disorders in medication trials. We conducted a secondary analysis on the Aberrant Behavior Checklist Social Withdrawal subscale using data from two federally-funded, multi-site, randomized trials with risperidone. Study 1 included 52 subjects assigned to placebo and 49 subjects to risperidone under double-blind conditions. Study 2 included 49 subjects assigned to risperidone only and 75 subjects assigned to risperidone plus parent training. After 8 weeks of treatment, all active treatments were superior to placebo (effect sizes ranging from 0.42 to 0.65). The findings suggest that the Social Withdrawal subscale may be a useful measure of social disability in acute treatment trials. En ligne : http://dx.doi.org/10.1007/s10803-012-1689-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=192 [article] Brief Report: Social Disability in Autism Spectrum Disorder: Results from Research Units on Pediatric Psychopharmacology (RUPP) Autism Network Trials [texte imprimé] / Lawrence SCAHILL, Auteur ; Victoria HALLETT, Auteur ; Michael G. AMAN, Auteur ; Christopher J. MCDOUGLE, Auteur ; L. Eugene ARNOLD, Auteur ; James T. MCCRACKEN, Auteur ; Elaine TIERNEY, Auteur ; Yanhong DENG, Auteur ; James DZIURA, Auteur ; Benedetto VITIELLO, Auteur . - p.739-746. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 43-3 (March 2013) . - p.739-746 Mots-clés : Social disability  Autism  Research Units on Pediatric Psychopharmacology (RUPP)  Autism Network  Social withdrawal  Risperidone  Aberrant Behavior Checklist Index. décimale : PER Périodiques Résumé : There is growing interest in measuring social disability as a core element of autism spectrum disorders in medication trials. We conducted a secondary analysis on the Aberrant Behavior Checklist Social Withdrawal subscale using data from two federally-funded, multi-site, randomized trials with risperidone. Study 1 included 52 subjects assigned to placebo and 49 subjects to risperidone under double-blind conditions. Study 2 included 49 subjects assigned to risperidone only and 75 subjects assigned to risperidone plus parent training. After 8 weeks of treatment, all active treatments were superior to placebo (effect sizes ranging from 0.42 to 0.65). The findings suggest that the Social Withdrawal subscale may be a useful measure of social disability in acute treatment trials. En ligne : http://dx.doi.org/10.1007/s10803-012-1689-3 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=192

Cholesterol Deficit in Autism: Insights from Smith–Lemli–Opitz Syndrome / Alka ANEJA

Public ISBD contenu dans Autism / Andrew W. ZIMMERMAN Titre : Cholesterol Deficit in Autism: Insights from Smith–Lemli–Opitz Syndrome Type de document : texte imprimé Auteurs : Alka ANEJA, Auteur ; Elaine TIERNEY, Auteur Année de publication : 2008 Importance : p.69-79 Langues : Anglais (eng) Mots-clés : Cholesterole  Syndrome de Smith-Lemli-Opitz  Neurostéroide Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=703 contenu dans Autism / Andrew W. ZIMMERMAN Cholesterol Deficit in Autism: Insights from Smith–Lemli–Opitz Syndrome [texte imprimé] / Alka ANEJA, Auteur ; Elaine TIERNEY, Auteur . - 2008 . - p.69-79. Langues : Anglais (eng) Mots-clés : Cholesterole  Syndrome de Smith-Lemli-Opitz  Neurostéroide Index. décimale : AUT-B AUT-B - L'Autisme - Ouvrages généraux et scientifiques Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=703

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Development, behavior, and biomarker characterization of Smith-Lemli-Opitz syndrome: an update / Audrey THURM in Journal of Neurodevelopmental Disorders, 8-1 (December 2016)  

Public ISBD [article]  inJournal of Neurodevelopmental Disorders > 8-1 (December 2016) . - p.12 Titre : Development, behavior, and biomarker characterization of Smith-Lemli-Opitz syndrome: an update Type de document : texte imprimé Auteurs : Audrey THURM, Auteur ; Elaine TIERNEY, Auteur ; Cristan FARMER, Auteur ; Phebe ALBERT, Auteur ; Lisa JOSEPH, Auteur ; Susan E. SWEDO, Auteur ; Simona BIANCONI, Auteur ; Irena BUKELIS, Auteur ; Courtney WHEELER, Auteur ; Geeta SARPHARE, Auteur ; Diane LANHAM, Auteur ; Christopher A. WASSIF, Auteur ; Forbes D. PORTER, Auteur Article en page(s) : p.12 Langues : Anglais (eng) Mots-clés : Autism  Developmental delay  Smith-Lemli-Opitz  Sterols Index. décimale : PER Périodiques Résumé : BACKGROUND: Smith-Lemli-Opitz syndrome (SLOS) is an autosomal recessive inborn error of cholesterol metabolism syndrome with neurocognitive manifestations. SLOS is the result of mutations in the gene encoding the 7-dehydrocholesterol reductase, which results in the elevation of the cholesterol precursor 7-dehydrocholesterol (7-DHC). Previous reports indicate that intellectual disability, behavioral disturbances, and autism symptoms are frequently part of the SLOS behavioral phenotype. In the current study, we characterize the developmental history and current behavior of 33 individuals with SLOS aged 4 to 23 years and report on biomarkers 7-DHC and 8-DHC in relation to cognition and behavior. METHODS: This was an observational case series, wherein participants with SLOS underwent extensive behavioral evaluation of cognitive function, adaptive function, autism symptoms, and problem behaviors, in addition to parent report of developmental milestones. Serum and CSF were contemporaneously obtained from the majority of participants. RESULTS: Developmental milestones such as walking, talking, and toileting were uniformly delayed. Overall levels of cognitive and adaptive functioning were low; no participant received adaptive behavior scores in the average range, and the mean level of cognitive functioning in the full sample was in the moderate range of impairment. Aggressive behavior was present in nearly half of participants. Although the majority of participants had elevated scores on the gold standard autism diagnostic instruments, only about half of participants received a clinical diagnosis of autism spectrum disorder. Finally, while CSF cholesterol was not found to correlate with cognitive or adaptive functioning, both serum and CSF 7-DHC and 8-DHC (and their ratios with cholesterol) were moderately and negatively correlated with functioning in this group. CONCLUSIONS: A history of developmental delay, followed by intellectual disability, is common in individuals with SLOS. Although autism spectrum disorder appears to be a frequent diagnosis in this population, it is apparent that the low level of functioning observed in SLOS may artificially inflate scores on standard autism assessments. Our findings further support that cholesterol precursors 7-DHC and 8-DHC are important biomarkers of the level of functioning in SLOS, especially regarding cognitive abilities, and thus may be to explore as mediators within the context of treatment trials. TRIAL REGISTRATION: ClinicalTrials.gov, NCT00001721, NCT00064792. En ligne : http://dx.doi.org/10.1186/s11689-016-9145-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=348 [article] Development, behavior, and biomarker characterization of Smith-Lemli-Opitz syndrome: an update [texte imprimé] / Audrey THURM, Auteur ; Elaine TIERNEY, Auteur ; Cristan FARMER, Auteur ; Phebe ALBERT, Auteur ; Lisa JOSEPH, Auteur ; Susan E. SWEDO, Auteur ; Simona BIANCONI, Auteur ; Irena BUKELIS, Auteur ; Courtney WHEELER, Auteur ; Geeta SARPHARE, Auteur ; Diane LANHAM, Auteur ; Christopher A. WASSIF, Auteur ; Forbes D. PORTER, Auteur . - p.12. Langues : Anglais (eng) in Journal of Neurodevelopmental Disorders > 8-1 (December 2016) . - p.12 Mots-clés : Autism  Developmental delay  Smith-Lemli-Opitz  Sterols Index. décimale : PER Périodiques Résumé : BACKGROUND: Smith-Lemli-Opitz syndrome (SLOS) is an autosomal recessive inborn error of cholesterol metabolism syndrome with neurocognitive manifestations. SLOS is the result of mutations in the gene encoding the 7-dehydrocholesterol reductase, which results in the elevation of the cholesterol precursor 7-dehydrocholesterol (7-DHC). Previous reports indicate that intellectual disability, behavioral disturbances, and autism symptoms are frequently part of the SLOS behavioral phenotype. In the current study, we characterize the developmental history and current behavior of 33 individuals with SLOS aged 4 to 23 years and report on biomarkers 7-DHC and 8-DHC in relation to cognition and behavior. METHODS: This was an observational case series, wherein participants with SLOS underwent extensive behavioral evaluation of cognitive function, adaptive function, autism symptoms, and problem behaviors, in addition to parent report of developmental milestones. Serum and CSF were contemporaneously obtained from the majority of participants. RESULTS: Developmental milestones such as walking, talking, and toileting were uniformly delayed. Overall levels of cognitive and adaptive functioning were low; no participant received adaptive behavior scores in the average range, and the mean level of cognitive functioning in the full sample was in the moderate range of impairment. Aggressive behavior was present in nearly half of participants. Although the majority of participants had elevated scores on the gold standard autism diagnostic instruments, only about half of participants received a clinical diagnosis of autism spectrum disorder. Finally, while CSF cholesterol was not found to correlate with cognitive or adaptive functioning, both serum and CSF 7-DHC and 8-DHC (and their ratios with cholesterol) were moderately and negatively correlated with functioning in this group. CONCLUSIONS: A history of developmental delay, followed by intellectual disability, is common in individuals with SLOS. Although autism spectrum disorder appears to be a frequent diagnosis in this population, it is apparent that the low level of functioning observed in SLOS may artificially inflate scores on standard autism assessments. Our findings further support that cholesterol precursors 7-DHC and 8-DHC are important biomarkers of the level of functioning in SLOS, especially regarding cognitive abilities, and thus may be to explore as mediators within the context of treatment trials. TRIAL REGISTRATION: ClinicalTrials.gov, NCT00001721, NCT00064792. En ligne : http://dx.doi.org/10.1186/s11689-016-9145-x Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=348

Exploring the Manifestations of Anxiety in Children with Autism Spectrum Disorders / Victoria HALLETT in Journal of Autism and Developmental Disorders, 43-10 (October 2013)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 43-10 (October 2013) . - p.2341-2352 Titre : Exploring the Manifestations of Anxiety in Children with Autism Spectrum Disorders Type de document : texte imprimé Auteurs : Victoria HALLETT, Auteur ; Luc LECAVALIER, Auteur ; Denis G. SUKHODOLSKY, Auteur ; Noreen CIPRIANO, Auteur ; Michael G. AMAN, Auteur ; James T. MCCRACKEN, Auteur ; Christopher J. MCDOUGLE, Auteur ; Elaine TIERNEY, Auteur ; Bryan H. KING, Auteur ; Eric HOLLANDER, Auteur ; Linmarie SIKICH, Auteur ; Joel D. BREGMAN, Auteur ; Evdokia ANAGNOSTOU, Auteur ; Craig DONNELLY, Auteur ; Lily KATSOVICH, Auteur ; Kimberly DUKES, Auteur ; Benedetto VITIELLO, Auteur ; Kenneth D. GADOW, Auteur ; Lawrence SCAHILL, Auteur Article en page(s) : p.2341-2352 Langues : Anglais (eng) Mots-clés : Autism spectrum disorders  Anxiety  Measurement  Clinical Trials Index. décimale : PER Périodiques Résumé : This study explores the manifestation and measurement of anxiety symptoms in 415 children with ASDs on a 20-item, parent-rated, DSM-IV referenced anxiety scale. In both high and low-functioning children (IQ above vs. below 70), commonly endorsed items assessed restlessness, tension and sleep difficulties. Items requiring verbal expression of worry by the child were rarely endorsed. Higher anxiety was associated with functional language, IQ above 70 and higher scores on several other behavioral measures. Four underlying factors emerged: Generalized Anxiety, Separation Anxiety, Social Anxiety and Over-arousal. Our findings extend our understanding of anxiety across IQ in ASD and provide guidance for improving anxiety outcome measurement. En ligne : http://dx.doi.org/10.1007/s10803-013-1775-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=215 [article] Exploring the Manifestations of Anxiety in Children with Autism Spectrum Disorders [texte imprimé] / Victoria HALLETT, Auteur ; Luc LECAVALIER, Auteur ; Denis G. SUKHODOLSKY, Auteur ; Noreen CIPRIANO, Auteur ; Michael G. AMAN, Auteur ; James T. MCCRACKEN, Auteur ; Christopher J. MCDOUGLE, Auteur ; Elaine TIERNEY, Auteur ; Bryan H. KING, Auteur ; Eric HOLLANDER, Auteur ; Linmarie SIKICH, Auteur ; Joel D. BREGMAN, Auteur ; Evdokia ANAGNOSTOU, Auteur ; Craig DONNELLY, Auteur ; Lily KATSOVICH, Auteur ; Kimberly DUKES, Auteur ; Benedetto VITIELLO, Auteur ; Kenneth D. GADOW, Auteur ; Lawrence SCAHILL, Auteur . - p.2341-2352. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 43-10 (October 2013) . - p.2341-2352 Mots-clés : Autism spectrum disorders  Anxiety  Measurement  Clinical Trials Index. décimale : PER Périodiques Résumé : This study explores the manifestation and measurement of anxiety symptoms in 415 children with ASDs on a 20-item, parent-rated, DSM-IV referenced anxiety scale. In both high and low-functioning children (IQ above vs. below 70), commonly endorsed items assessed restlessness, tension and sleep difficulties. Items requiring verbal expression of worry by the child were rarely endorsed. Higher anxiety was associated with functional language, IQ above 70 and higher scores on several other behavioral measures. Four underlying factors emerged: Generalized Anxiety, Separation Anxiety, Social Anxiety and Over-arousal. Our findings extend our understanding of anxiety across IQ in ASD and provide guidance for improving anxiety outcome measurement. En ligne : http://dx.doi.org/10.1007/s10803-013-1775-1 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=215

Gastrointestinal Symptoms in a Sample of Children with Pervasive Developmental Disorders / Roumen N. NIKOLOV in Journal of Autism and Developmental Disorders, 39-3 (March 2009)  

Public ISBD [article]  inJournal of Autism and Developmental Disorders > 39-3 (March 2009) . - p.405-413 Titre : Gastrointestinal Symptoms in a Sample of Children with Pervasive Developmental Disorders Type de document : texte imprimé Auteurs : Roumen N. NIKOLOV, Auteur ; Lawrence SCAHILL, Auteur ; Bhavik SHAH, Auteur ; Maria RODOWSKI, Auteur ; Craig ERICKSON, Auteur ; Jelle LETTINGA, Auteur ; Karen E. BEARSS, Auteur ; David J. POSEY, Auteur ; Louise RITZ, Auteur ; Elaine TIERNEY, Auteur ; Christopher J. MCDOUGLE, Auteur ; Michael G. AMAN, Auteur ; Benedetto VITIELLO, Auteur ; L. Eugene ARNOLD, Auteur ; James T. MCCRACKEN, Auteur Année de publication : 2009 Article en page(s) : p.405-413 Langues : Anglais (eng) Mots-clés : Autism Chronic-gastrointestinal-problems Pervasive-developmental-disorders Index. décimale : PER Périodiques Résumé : Objective To evaluate gastrointestinal (GI) problems in a large, well-characterized sample of children with pervasive developmental disorders (PDDs). Methods One hundred seventy two children entering one of two trials conducted by the Research Units on Pediatric Psychopharmacology (RUPP) Autism Network were assessed comprehensively prior to starting treatment and classified with regard to GI symptoms. Results Thirty nine (22.7%) were positive for GI problems, primarily constipation and diarrhea. Those with GI problems were no different from subjects without GI problems in demographic characteristics, measures of adaptive functioning, or autism symptom severity. Compared to children without GI problems, those with GI problems showed greater symptom severity on measures of irritability, anxiety, and social withdrawal. Those with GI problems were also less likely to respond to treatment. En ligne : http://dx.doi.org/10.1007/s10803-008-0637-8 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=695 [article] Gastrointestinal Symptoms in a Sample of Children with Pervasive Developmental Disorders [texte imprimé] / Roumen N. NIKOLOV, Auteur ; Lawrence SCAHILL, Auteur ; Bhavik SHAH, Auteur ; Maria RODOWSKI, Auteur ; Craig ERICKSON, Auteur ; Jelle LETTINGA, Auteur ; Karen E. BEARSS, Auteur ; David J. POSEY, Auteur ; Louise RITZ, Auteur ; Elaine TIERNEY, Auteur ; Christopher J. MCDOUGLE, Auteur ; Michael G. AMAN, Auteur ; Benedetto VITIELLO, Auteur ; L. Eugene ARNOLD, Auteur ; James T. MCCRACKEN, Auteur . - 2009 . - p.405-413. Langues : Anglais (eng) in Journal of Autism and Developmental Disorders > 39-3 (March 2009) . - p.405-413 Mots-clés : Autism Chronic-gastrointestinal-problems Pervasive-developmental-disorders Index. décimale : PER Périodiques Résumé : Objective To evaluate gastrointestinal (GI) problems in a large, well-characterized sample of children with pervasive developmental disorders (PDDs). Methods One hundred seventy two children entering one of two trials conducted by the Research Units on Pediatric Psychopharmacology (RUPP) Autism Network were assessed comprehensively prior to starting treatment and classified with regard to GI symptoms. Results Thirty nine (22.7%) were positive for GI problems, primarily constipation and diarrhea. Those with GI problems were no different from subjects without GI problems in demographic characteristics, measures of adaptive functioning, or autism symptom severity. Compared to children without GI problems, those with GI problems showed greater symptom severity on measures of irritability, anxiety, and social withdrawal. Those with GI problems were also less likely to respond to treatment. En ligne : http://dx.doi.org/10.1007/s10803-008-0637-8 Permalink : https://www.cra-rhone-alpes.org/cid/opac_css/index.php?lvl=notice_display&id=695

Hypothesis: The Role of Sterols in Autism Spectrum Disorder / Ryan W.Y. LEE in Autism Research and Treatment, (February 2011)  

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Positive Effects of Methylphenidate on Social Communication and Self-Regulation in Children with Pervasive Developmental Disorders and Hyperactivity / Laudan B. JAHROMI in Journal of Autism and Developmental Disorders, 39-3 (March 2009)  

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Sensitivity of the modified Children’s Yale–Brown Obsessive Compulsive Scale to detect change: Results from two multi-site trials / Lawrence SCAHILL in Autism, 20-2 (February 2016)  

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Smith-Lemli-Opitz Syndrome and Role of Cholesterol in Autism / Geeta SARPHARE

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