title: Luteoma of pregnancy associated with nearly complete virilization of genetically female twins.
abstract: OBJECTIVE
  To describe a pregnancy that was complicated by the virilization of the mother and two 46XX infants.
  
  
  METHODS
  We outline the clinical presentation and diagnosis of the virilization of a mother and her twins, reviewing pertinent literature.
  
  
  RESULTS
  We report the case of a 40-year-old Caucasian female who conceived a trichorionic triplet pregnancy through in vitro fertilization (IVF) but underwent cytoreduction at 13 weeks of gestation, leaving a diamniotic dichorionic twin pregnancy. At 16 weeks of gestation the mother experienced increasing acne, facial hair, and deepening of her voice. Due to preeclampsia, the twins were delivered via caesarean section at 33 weeks of gestation. The infants had male-appearing external genitalia (Prader score IV-V) but no palpable gonads. Congenital adrenal hyperplasia was ruled out for both twins and they were both found to have a uterus and a 46XX karyotype. Maternal testosterone level was elevated at birth (1,981 ng/dL), but the infants had normal levels. Maternal testosterone levels returned to normal after delivery, consistent with a luteoma of pregnancy, although imaging was negative for a mass.
  
  
  CONCLUSIONS
  This is the second reported case of complete virilization associated with a luteoma of pregnancy. Whether or not IVF and related procedures increase the risk for a luteoma and whether or not fetal reduction procedures disrupt placental aromatases and increase the risk of virilization in the face of elevated androgen levels are questions that require further research.
authors.affiliation: Department of Pediatrics, Baystate Medical Center, Springfield, Massachusetts.
authors.forename: Thomas L
authors.initials: TL
authors.lastname: Wadzinski
authors.affiliation: Department of Internal Medicine, Baystate Medical Center, Springfield, Massachusetts.
authors.forename: Yousef
authors.initials: Y
authors.lastname: Altowaireb
authors.affiliation: Department of Pediatrics, Baystate Medical Center, Springfield, Massachusetts Department of Internal Medicine, Baystate Medical Center, Springfield, Massachusetts.
authors.forename: Rashim
authors.initials: R
authors.lastname: Gupta
authors.affiliation: Department of Pediatrics, Baystate Medical Center, Springfield, Massachusetts.
authors.forename: Rushika
authors.initials: R
authors.lastname: Conroy
authors.affiliation: Department of Internal Medicine, Baystate Medical Center, Springfield, Massachusetts.
authors.forename: Kamal
authors.initials: K
authors.lastname: Shoukri