• Media type: E-Article
  • Title: Recurrent Intragenic Duplication within the NR5A1 Gene and Severe Proximal Hypospadias
  • Contributor: Peycelon, Matthieu; Mansour-Hendili, Lamisse; Hyon, Capucine; Collot, Nathalie; Houang, Muriel; Legendre, Marie; Chabaud, Maud; Bouvier, Marie-Dominique; Audry, Georges; Amselem, Serge; Siffroi, Jean-Pierre
  • Published: S. Karger AG, 2017
  • Published in: Sexual Development, 11 (2017) 5-6, Seite 293-297
  • Language: English
  • DOI: 10.1159/000485909
  • ISSN: 1661-5425; 1661-5433
  • Origination:
  • Footnote:
  • Description: A heterozygous intragenic duplication within the repeated area (CTGCAGCTG)×2 of the <i>NR5A1</i> gene was found in a 15-year-old 46,XY DSD (disorders/differences of sex development) patient with micropenis and severe proximal hypospadias. This heterozygous duplication has already been described twice in boys with a similar phenotype, whereas a deletion of 3 amino acids at the same position in the protein SF-1 has been described in a 46,XX patient with primary ovarian failure and short stature. These data suggest that this region within the <i>NR5A1</i> gene has an important role for SF-1 protein function in gonads and is a hotspot for intragenic rearrangements.