Literature DB >> 11938445

Do reduced levels of steroid 21-hydroxylase confer a survival advantage in fetuses affected by sex chromosome aberrations?

Vilma Mantovani1, Elisabetta Dondi, Daniela Larizza, Mariangela Cisternino, Michela Bragliani, Mariagabriella Viggiani, Miryam Martinetti, Mariaclara Cuccia.   

Abstract

We investigated whether molecular defects in the CYP21 gene were detectable in two common sex chromosome aberrations, the Turner and the Klinefelter syndromes. We found abnormal 17-hydroxyprogesterone levels after adrenal stimulation in 26/60 (43.3%) patients affected by these chromosome aberrations, as compared with only 11/68 (16.2%) normal controls (P=0.0014, odds ratio 4.0). Screening of the CYP21 gene identified a single Val281Leu missense mutation in exon 7 in 9/63 (14.3%) of the patients, all nine of whom were heterozygote carriers; the mutation frequency was significantly higher than in the general population (P=0.007, odds ratio=3.5). The hormonal and molecular data indicate that these common sex chromosome aberrations are associated with a remarkably high frequency of steroidogenic defects. It may be hypothesised that reduced levels of steroid 21-hydroxylase could confer a survival advantage, leading to a successful pregnancy.

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Year:  2002        PMID: 11938445     DOI: 10.1038/sj.ejhg.5200778

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  3 in total

1.  Premature pubarche in a child with abnormal 3β-hydroxysteroid dehydrogenase function and Klinefelter syndrome: the intriguing relationship between androgen deficiency and excess.

Authors:  Michele Gortakowski; Rushika Conroy; Luisa Aguiar; Holley Allen
Journal:  Clin Case Rep       Date:  2016-12-20

2.  Revisiting the prevalence of nonclassic congenital adrenal hyperplasia in US Ashkenazi Jews and Caucasians.

Authors:  Fady Hannah-Shmouni; Rachel Morissette; Ninet Sinaii; Meredith Elman; Toni R Prezant; Wuyan Chen; Ann Pulver; Deborah P Merke
Journal:  Genet Med       Date:  2017-05-25       Impact factor: 8.822

3.  A rare combination: congenital adrenal hyperplasia due to 21 hydroxylase deficiency and Turner syndrome.

Authors:  Havva Nur Peltek Kendirci; Zehra Aycan; Semra Çetinkaya; Veysel Nijat Baş; Sebahat Yılmaz Ağladıoğlu; Aşan Önder
Journal:  J Clin Res Pediatr Endocrinol       Date:  2012-12
  3 in total

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