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Pregnancy in a hermaphrodite with a male-predominant mosaic karyotype.

AbstractOBJECTIVE:
To report a pregnancy in a hermaphrodite and review of the literature.
DESIGN:
Case report and literature review.
SETTING:
Clinical.
PATIENT(S):
A patient with male-predominant mosaic karyotype 96% 46XY.
INTERVENTION(S):
Removal of left ovotestis in combination with a supracervical hysterectomy and bilateral salpingo-oophorectomy.
MAIN OUTCOME MEASURE(S):
Identification of published cases of pregnancy and hermaphroditism.
RESULT(S):
The current patient had previous pregnancy and a wedge biopsy of her left gonad, which demonstrated an ovotestis and an area suspicious for a gonadoblastoma. After delivery of her second pregnancy, the patient underwent a hysterectomy and bilateral salpingo-oophorectomy. The histopathologic evaluation following bilateral oophorectomy demonstrated a residual ovotestis without further evidence of a gonadoblastoma. Review of the literature identified 10 other cases of pregnancy in a hermaphrodite patient.
CONCLUSION(S):
This is the 11th reported case of fertility in a true hermaphrodite and only the second report of a mosaic true hermaphrodite to demonstrate fertility. This is the only case of a pregnancy involving a male-predominant mosaic 96% 46XY and the only case to confirm the genetics of the offspring.
AuthorsSamantha A Schoenhaus, Scott E Lentz, Peter Saber, Malcom G Munro, Seth Kivnick
JournalFertility and sterility (Fertil Steril) Vol. 90 Issue 5 Pg. 2016.e7-10 (Nov 2008) ISSN: 1556-5653 [Electronic] United States
PMID18394621 (Publication Type: Case Reports, Journal Article, Review)
Topics
  • Adult
  • Female
  • Fertility
  • Gonadal Dysgenesis, Mixed (genetics, pathology, physiopathology)
  • Gonadoblastoma (genetics, pathology, prevention & control)
  • Humans
  • Hysterectomy
  • Karyotyping
  • Laparoscopy
  • Live Birth
  • Male
  • Mosaicism
  • Ovarian Neoplasms (genetics, pathology, prevention & control)
  • Ovariectomy
  • Ovary (pathology, surgery)
  • Ovotesticular Disorders of Sex Development (genetics, pathology, physiopathology)
  • Pregnancy
  • Testicular Neoplasms (genetics, pathology, prevention & control)
  • Testis (pathology, surgery)

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