Pregnancy in a hermaphrodite with a male-predominant mosaic karyotype
Received 5 January 2008; received in revised form 25 January 2008; accepted 31 January 2008. published online 04 April 2008.
Objective
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.
Kaiser Permanente Southern California, West Los Angeles and Los Angeles Medical Centers, Los Angeles, California
Reprint requests: Seth Kivnick, M.D., Kaiser Permanente, 6041 Cadillac Blvd., Los Angeles, CA 90034 (FAX: 323-857-3946).
S.A.S. has nothing to disclose. S.E.L. has nothing to disclose. P.S. has nothing to disclose. M.G.M. is a consultant for Ethicon Women's Health, Covidien, Gynesonics, Karl Storz Endoscopy, and AMAG Pharmaceuticals and is a shareholder in Gynesonics and Impres Medical. S.K. has nothing to disclose.