Literature DB >> 2018071

Heterozygous fragile X female: historical, physical, cognitive, and cytogenetic features.

A Cronister1, R Schreiner, M Wittenberger, K Amiri, K Harris, R J Hagerman.   

Abstract

Historical, physical, cognitive, and cytogenetic data were documented in 105 heterozygous fragile X [fra(X)] females and 90 controls in a prospective fashion. For comparisons, we divided heterozygotes and controls into those with cognitive impairment (IQ less than 85) and normal IQ (IQ greater than or equal to 85). The only finding that was significantly more frequent in impaired heterozygotes compared with impaired controls chi 2 analysis was shyness. Features that were more frequent in normal IQ heterozygotes compared with normal controls were voluntary thumb dislocation and hyperextensible metacarpal-phalangeal (MP) joints. Comparisons among heterozygotes demonstrated more math problems, hand biting, strabismus, high-arched palate, hyperextensible finger joints, and flat feet in impaired heterozygotes than in normal heterozygotes. Premature menopause was present in 8 of 61 normal heterozygotes and in none of the impaired heterozygotes. A multiple regression analysis demonstrated a significant inverse correlation between the percent fragility and IQ for the heterozygotes as a group. However, no correlation existed between IQ and fragility when the percent fragility was 2% or greater. However, a higher percentage of fragility was positively correlated with the total number of physical findings present.

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Year:  1991        PMID: 2018071     DOI: 10.1002/ajmg.1320380221

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


  51 in total

1.  Premature ovarian failure is associated with maternally and paternally inherited premutation in Brazilian families with fragile X.

Authors:  A M Vianna-Morgante; S S Costa
Journal:  Am J Hum Genet       Date:  2000-07       Impact factor: 11.025

2.  Premature ovarian failure among fragile X premutation carriers: parent-of-origin effect?

Authors:  S L Sherman
Journal:  Am J Hum Genet       Date:  2000-06-12       Impact factor: 11.025

Review 3.  Fragile X: leading the way for targeted treatments in autism.

Authors:  Lulu W Wang; Elizabeth Berry-Kravis; Randi J Hagerman
Journal:  Neurotherapeutics       Date:  2010-07       Impact factor: 7.620

Review 4.  Advances in the Understanding of the Gabaergic Neurobiology of FMR1 Expanded Alleles Leading to Targeted Treatments for Fragile X Spectrum Disorder.

Authors:  Reymundo Lozano; Veronica Martinez-Cerdeno; Randi J Hagerman
Journal:  Curr Pharm Des       Date:  2015       Impact factor: 3.116

Review 5.  Fragile X-associated tremor/ataxia syndrome (FXTAS): pathology and mechanisms.

Authors:  Paul Hagerman
Journal:  Acta Neuropathol       Date:  2013-06-21       Impact factor: 17.088

6.  Studies of FRAXA and FRAXE in women with premature ovarian failure.

Authors:  A Murray; J Webb; S Grimley; G Conway; P Jacobs
Journal:  J Med Genet       Date:  1998-08       Impact factor: 6.318

7.  Mental status of females with an FMR1 gene full mutation.

Authors:  B B de Vries; A M Wiegers; A P Smits; S Mohkamsing; H J Duivenvoorden; J P Fryns; L M Curfs; D J Halley; B A Oostra; A M van den Ouweland; M F Niermeijer
Journal:  Am J Hum Genet       Date:  1996-05       Impact factor: 11.025

8.  Fragile-X: neuropsychological test performance, CGG triplet repeat lengths, and hippocampal volumes.

Authors:  P Jäkälä; T Hänninen; M Ryynänen; M Laakso; K Partanen; A Mannermaa; H Soininen
Journal:  J Clin Invest       Date:  1997-07-15       Impact factor: 14.808

9.  Prevalence and phenotype consequence of FRAXA and FRAXE alleles in a large, ethnically diverse, special education-needs population.

Authors:  D C Crawford; K L Meadows; J L Newman; L F Taft; D L Pettay; L B Gold; S J Hersey; E F Hinkle; M L Stanfield; P Holmgreen; M Yeargin-Allsopp; C Boyle; S L Sherman
Journal:  Am J Hum Genet       Date:  1999-02       Impact factor: 11.025

10.  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

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