Literature DB >> 14505067

Autistic-like behaviour profile and psychiatric morbidity in Fragile X Syndrome: a prospective ten-year follow-up study.

Manga Sabaratnam1, Naga Venkatesha Murthy, Anusha Wijeratne, Ann Buckingham, Stewart Payne.   

Abstract

In subjects with Fragile X Syndrome (FXS), the mutation of Fragile X Mental Retardation Type 1 ( FMR-1) gene at Xq27.3 predisposes to Mental Retardation (MR), autistic-like behaviour and to a variety of psychiatric syndromes. However, the longitudinal course of autistic-like behaviour profile and psychiatric morbidity is untested. In this study, we followed up people with FXS for 10 years to establish the stability of their autistic- like behaviour profile and psychiatric morbidity. The autisticlike behaviour profile was assessed using Brief Disability Assessment Schedule (B-DAS) and relevant items from Handicaps, Behaviour and Skills (HBS) Schedule. The psychiatric morbidity was assessed using data from the case notes, Mini Psychiatric Assessment Schedule for Adults with Developmental Disability (Mini PAS-ADD) and clinical interview. Our findings suggest that the autistic-like behaviour pattern is a stable phenotypic feature of FXS, but for increase in resistance to change over time. There is a tenfold increase in the prevalence of psychiatric morbidity in FXS compared to the general population, which does not increase significantly over time.

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Year:  2003        PMID: 14505067     DOI: 10.1007/s00787-003-0333-3

Source DB:  PubMed          Journal:  Eur Child Adolesc Psychiatry        ISSN: 1018-8827            Impact factor:   4.785


  25 in total

1.  Change in the Behavioral Phenotype of Adolescents and Adults with FXS: Role of the Family Environment.

Authors:  Leann E Smith; Jinkuk Hong; Jan S Greenberg; Marsha R Mailick
Journal:  J Autism Dev Disord       Date:  2016-05

2.  Increased Exposure to Rigid Routines can Lead to Increased Challenging Behavior Following Changes to Those Routines.

Authors:  Leah E Bull; Chris Oliver; Eleanor Callaghan; Kate A Woodcock
Journal:  J Autism Dev Disord       Date:  2015-06

3.  Profiles of receptive and expressive language abilities in boys with comorbid fragile X syndrome and autism.

Authors:  Andrea McDuffie; Sara Kover; Leonard Abbeduto; Pamela Lewis; Ted Brown
Journal:  Am J Intellect Dev Disabil       Date:  2012-01

4.  Behavioral phenotype of fragile X syndrome in adolescence and adulthood.

Authors:  Leann E Smith; Erin T Barker; Marsha Mailick Seltzer; Leonard Abbeduto; Jan S Greenberg
Journal:  Am J Intellect Dev Disabil       Date:  2012-01

5.  Consistency between research and clinical diagnoses of autism among boys and girls with fragile X syndrome.

Authors:  J Klusek; G E Martin; M Losh
Journal:  J Intellect Disabil Res       Date:  2014-02-17

6.  Decreased home cage movement and oromotor impairments in adult Fmr1-KO mice.

Authors:  S J Bonasera; T R Chaudoin; E H Goulding; M Mittek; A Dunaevsky
Journal:  Genes Brain Behav       Date:  2017-03-29       Impact factor: 3.449

7.  Language Development in Individuals with Fragile X Syndrome.

Authors:  Lizbeth H Finestack; Erica K Richmond; Leonard Abbeduto
Journal:  Top Lang Disord       Date:  2009-04

Review 8.  Public Health Literature Review of Fragile X Syndrome.

Authors:  Melissa Raspa; Anne C Wheeler; Catharine Riley
Journal:  Pediatrics       Date:  2017-06       Impact factor: 7.124

9.  Autism spectrum disorder in fragile X syndrome: a longitudinal evaluation.

Authors:  R Nick Hernandez; Rachel L Feinberg; Rebecca Vaurio; Natalie M Passanante; Richard E Thompson; Walter E Kaufmann
Journal:  Am J Med Genet A       Date:  2009-06       Impact factor: 2.802

10.  Involvement of FMRP in Primary MicroRNA Processing via Enhancing Drosha Translation.

Authors:  Rui-Ping Wan; Lin-Tao Zhou; Hai-Xuan Yang; Yong-Ting Zhou; Shun-Hua Ye; Qi-Hua Zhao; Mei-Mei Gao; Wei-Ping Liao; Yong-Hong Yi; Yue-Sheng Long
Journal:  Mol Neurobiol       Date:  2016-03-19       Impact factor: 5.590

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