Literature DB >> 8844077

Molecular/clinical correlations in females with fragile X.

W E Sobesky1, A K Taylor, B F Pennington, L Bennetto, D Porter, J Riddle, R J Hagerman.   

Abstract

Females who are affected by fragile X syndrome (FXS) can have significant physical, neuropsychological and emotional involvement. This study was designed to explore the relationships between these three domains and to learn how the degree of involvement in each of these phenotypic areas relates to molecular parameters including CGG repeat length and activation ratio (the proportion of normal FMR1 alleles on the active X chromosome). Three groups of females were studied: 35 women who grew up in a fragile X family but do not carry an FMR1 mutation, 92 women with a premutation, and 29 women with a full mutation. Correlations between neurocognitive, physical and emotional traits were calculated for each of the three groups. Within the full mutation group significant correlations were seen between schizotypal traits and full scale IQ. The Lie scale was significantly correlated with the physical findings index. The activation ratio correlated significantly with the measure of executive function (r = .50, P = .01). There was a trend toward correlations of activation ratio with the physical index score, outer ear prominence and IQ. CGG repeat number significantly correlated only with the physical index (r = .44, P = .01). Thus, activation ratio may be the more pertinent molecular parameter in full mutation women in determining the degree of cognitive and physical phenotypic involvement.

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Year:  1996        PMID: 8844077     DOI: 10.1002/(SICI)1096-8628(19960809)64:2<340::AID-AJMG21>3.0.CO;2-E

Source DB:  PubMed          Journal:  Am J Med Genet        ISSN: 0148-7299


  14 in total

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2.  Social cognition in adolescent girls with fragile x syndrome.

Authors:  Lyn S Turkstra; Leonard Abbeduto; Peter Meulenbroek
Journal:  Am J Intellect Dev Disabil       Date:  2014-07

Review 3.  Fragile X syndrome. Molecular and clinical insights and treatment issues.

Authors:  R J Hagerman
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Review 4.  Fragile X-associated tremor/ataxia syndrome.

Authors:  Paul J Hagerman; Randi J Hagerman
Journal:  Ann N Y Acad Sci       Date:  2015-01-26       Impact factor: 5.691

Review 5.  Neuropsychiatric symptoms of fragile X syndrome: pathophysiology and pharmacotherapy.

Authors:  John A Tsiouris; W Ted Brown
Journal:  CNS Drugs       Date:  2004       Impact factor: 5.749

6.  Psychological symptoms correlate with reduced hippocampal volume in fragile X premutation carriers.

Authors:  P E Adams; J S Adams; D V Nguyen; D Hessl; J A Brunberg; F Tassone; W Zhang; K Koldewyn; S M Rivera; J Grigsby; L Zhang; C Decarli; P J Hagerman; R J Hagerman
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2010-04-05       Impact factor: 3.568

7.  Expanded clinical phenotype of women with the FMR1 premutation.

Authors:  Sarah M Coffey; Kylee Cook; Nicole Tartaglia; Flora Tassone; Danh V Nguyen; Ruiqin Pan; Hannah E Bronsky; Jennifer Yuhas; Mariya Borodyanskaya; Jim Grigsby; Melanie Doerflinger; Paul J Hagerman; Randi J Hagerman
Journal:  Am J Med Genet A       Date:  2008-04-15       Impact factor: 2.802

8.  Recommendations from multi-disciplinary focus groups on cascade testing and genetic counseling for fragile X-associated disorders.

Authors:  Allyn McConkie-Rosell; Liane Abrams; Brenda Finucane; Amy Cronister; Louise W Gane; Sarah M Coffey; Stephanie Sherman; Lawrence M Nelson; Elizabeth Berry-Kravis; David Hessl; Sufen Chiu; Natalie Street; Ajay Vatave; Randi J Hagerman
Journal:  J Genet Couns       Date:  2007-05-12       Impact factor: 2.537

Review 9.  Fragile X-associated tremor/ataxia syndrome: clinical phenotype, diagnosis, and treatment.

Authors:  Maureen A Leehey
Journal:  J Investig Med       Date:  2009-12       Impact factor: 2.895

10.  Investigation of phenotypes associated with mood and anxiety among male and female fragile X premutation carriers.

Authors:  Jessica Ezzell Hunter; Emily Graves Allen; Ann Abramowitz; Michele Rusin; Mary Leslie; Gloria Novak; Debra Hamilton; Lisa Shubeck; Krista Charen; Stephanie L Sherman
Journal:  Behav Genet       Date:  2008-06-06       Impact factor: 2.805

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