[Swyer syndrome: report of 5 cases]

J Gynecol Obstet Biol Reprod (Paris). 2009 May;38(3):220-5. doi: 10.1016/j.jgyn.2007.12.004. Epub 2008 Feb 1.
[Article in French]

Abstract

Introduction: Swyer's syndrome is a distinct type of pure gonadal dysgenesis characterized by a 46 XY karyotype in female phenotypic patients. It shows an abnormality in testicular differentiation. The disease is a sex-reversal disorder resulting from embryonic testicular regression sequences.

Material and methods: We present the clinical, sonographic and endocrine findings in five cases of phenotypic young girls with XY karyotype and gonadal dysgenesis. The treatment and the follow-up are discussed.

Results: The mean age was of 17.6 years. All patients presented with primary amenorrhea. All patients had female-type external genitalia. Secondary sexual characters were merely developed in all cases. FSH levels were high with a mean of 80.5. The surgical findings were steak fibrous gonads, two cases of gonadoblastoma and one case of dysgerminoma in a 13-year-old teenager.

Conclusion: The risk of gonadal neoplasia is high, dictating early prophylactic removal of these dysgenesic gonads.

Publication types

  • Case Reports
  • English Abstract

MeSH terms

  • Adolescent
  • Amenorrhea / genetics
  • Dysgerminoma / diagnosis
  • Dysgerminoma / surgery
  • Female
  • Follicle Stimulating Hormone / blood
  • Gonadal Dysgenesis, 46,XY / diagnosis*
  • Gonadal Dysgenesis, 46,XY / surgery*
  • Gonadoblastoma / diagnosis
  • Gonadoblastoma / surgery
  • Humans
  • Ovarian Neoplasms / diagnosis
  • Ovarian Neoplasms / surgery
  • Ovary / abnormalities
  • Uterus / abnormalities
  • Young Adult

Substances

  • Follicle Stimulating Hormone