| Effect of the deficits of fragile X mental retardation protein on cognitive status of fragile x males and females assessed by robust pedigree analysis. | |
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MedLine Citation:
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PMID: 12476071 Owner: NLM Status: MEDLINE |
Abstract/OtherAbstract:
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The effect of the fragile X mental retardation 1 (FMR1) gene product (fragile X mental retardation protein [FMRP]) deficits on Full-Scale IQ (FSIQ) and FSIQ-adjusted Wechsler subtests and index scores in fragile X disorder were assessed using a robust modification of the maximum likelihood estimators for pedigree data. The results from 144 extended families have demonstrated a linear effect of progressively reduced levels of FMRP on the FSIQ and all subtest and summary scores in either gender. The effect of FMRP in decreasing FSIQ-adjusted subtest scores was highly significant for Digit Span, Symbol Search, Object Assembly, and Picture Arrangement, with a consistent trend in both genders. Heritability for FSIQ and unadjusted subtest scores estimated from the covariance model did not exceed 50% and varied widely from the highest for Verbal score to the lowest for Picture Completion score. Possible mechanisms by which FMRP deficit impacts on specific weaknesses in fragile X are considered on the basis of present data. |
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Authors:
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Danuta Z Loesch; Richard M Huggins; Quang M Bui; Jennifer L Epstein; Annette K Taylor; Randi Jensen Hagerman |
Publication Detail:
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Type: Journal Article; Research Support, U.S. Gov't, P.H.S. |
Journal Detail:
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Title: Journal of developmental and behavioral pediatrics : JDBP Volume: 23 ISSN: 0196-206X ISO Abbreviation: J Dev Behav Pediatr Publication Date: 2002 Dec |
Date Detail:
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Created Date: 2002-12-11 Completed Date: 2003-04-15 Revised Date: 2007-11-14 |
Medline Journal Info:
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Nlm Unique ID: 8006933 Medline TA: J Dev Behav Pediatr Country: United States |
Other Details:
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Languages: eng Pagination: 416-23 Citation Subset: IM |
Affiliation:
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School of Psychological Science, La Trobe University, Melbourne. d. loesch@latrobe.edu.au |
Export Citation:
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| MeSH Terms | |
Descriptor/Qualifier:
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Adolescent Adult Aged Child Child, Preschool Cognition Disorders / diagnosis Female Fragile X Mental Retardation Protein Fragile X Syndrome / genetics* Humans Male Mental Retardation / genetics* Middle Aged Models, Biological Nerve Tissue Proteins / genetics* Pedigree RNA-Binding Proteins* Wechsler Scales |
| Grant Support | |
ID/Acronym/Agency:
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5M01RR00069/RR/NCRR NIH HHS; HD36071/HD/NICHD NIH HHS |
| Chemical | |
Reg. No./Substance:
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0/FMR1 protein, human; 0/Nerve Tissue Proteins; 0/RNA-Binding Proteins; 139135-51-6/Fragile X Mental Retardation Protein |
From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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