MATERNAL PREGNANCY LUTEOMA

A RARE CAUSE OF VIRILISATION IN A FEMALE NEWBORN AND MOTHER

Abstract

INTRODUCTION/BACKGROUND
Virilisation of a female newborn is commonly attributed to congenital adrenal hyperplasia but there are rarer causes that can be maternal in origin. Luteomas, a rare, benign androgen-producing ovarian tumour arising during pregnancy can result in both maternal and fetal virilization.

CASE
We describe a case of a newborn with ambiguous genitalia. This baby was born at 36 weeks via caesarean section for poor progress, weighing 2.8 kilograms at birth. Examination at birth revealed a prominent clitorophallic structure, fused labioscrotal folds but no palpable gonads. Otherwise, on general examination, there were no dysmorphic features or hyperpigmentation and serum electrolytes were normal with no hypoglycaemic episodes. On further assessment, 17 Hydroxyprogesterone (17-OHP) level was not elevated; karyotyping and radiological findings were consistent with a female gender. In hindsight, the mother recollected having signs of virilization, i.e., acneiform eruption on her upper chest and back, hirsutism, and deepening voice since the second trimester. Bilateral unhealthy, friable ovarian tumours were revealed intra-operatively which ruptured on handling. As the nature of the tumours was suspicious of malignancy, bilateral oophorectomy was done. Maternal beta human chorionic gonadotrophin (b-HCG) and alpha-fetoprotein (AFP) levels were elevated. The histopathological examination of the ovarian mass confirmed the diagnosis of pregnancy luteoma.

CONCLUSION
This case attests to the fact that rare causes of virilisation in a female baby cannot be overlooked. We thus need to be vigilant and have a high index of suspicion of maternal pregnancy luteomas as a possible cause of virilisation in a female baby.