Mantovani Vilma, Dondi Elisabetta, Larizza Daniela, Cisternino Mariangela, Bragliani Michela, Viggiani Mariagabriella, Martinetti Miryam, Cuccia Mariaclara
Laboratorio Centralizzato Settore Genetica, Policlinico S.Orsola-Malpighi, Bologna, Italy.
Eur J Hum Genet. 2002 Feb;10(2):137-40. doi: 10.1038/sj.ejhg.5200778.
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.