Complete gonadal dysgenesis 46,XY (Swyer syndrome) in two sisters and their mother's maternal aunt with a female phenotype

Fertil Steril. 2011 Apr;95(5):1786.e1-3. doi: 10.1016/j.fertnstert.2010.11.034. Epub 2010 Dec 9.

Abstract

Objective: To present a familial case of Swyer syndrome.

Design: Case report.

Setting: Academic medical center.

Patient(s): Two sisters with a main complaint of primary amenorrhea and another case, their mother's maternal aunt with the same history of primary amenorrhea but married with no consanguinity and no children.

Intervention(s): None.

Main outcome measure(s): The patients were studied from clinical, endocrinologic, and genetic perspectives.

Result(s): Chromosome analyses revealed a 46,XY male karyotype with no detectable mosaicism in both sisters and their mother's maternal aunt. Molecular studies of sex-determining region Y and molecular investigation undertaken for the two sisters revealed SRY negativity.

Conclusion(s): Gonadal dysgenesis can also be inherited as an X-linked disorder, and evidence exists from familial studies of perhaps autosomal inheritance.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Adult
  • Amenorrhea / diagnosis
  • Amenorrhea / etiology
  • Amenorrhea / genetics
  • Family*
  • Female
  • Gonadal Dysgenesis, 46,XY / complications
  • Gonadal Dysgenesis, 46,XY / diagnosis*
  • Gonadal Dysgenesis, 46,XY / genetics
  • Humans
  • Male
  • Mothers
  • Pedigree
  • Phenotype
  • Sex
  • Siblings*