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Published on: October 20, 2019
Pregnancy in a hermaphrodite with a male-predominant mosaic karyotype
Samantha A Schoenhaus1, Scott E Lentz, Peter Saber
1Kaiser Permanente Southern California, West Los Angeles and Los Angeles Medical Centers, Los Angeles, California 90034, USA.
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.
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