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Atypical case of preterm ovarian hyperstimulation syndrome

Michelle A Durst1, Brandy Wicklow2,3, Michael Narvey3,4

  • 1University of Manitoba, Department of Paediatrics and Child Health, Winnipeg, Manitoba, Canada.

BMJ Case Reports
|February 24, 2017
PubMed

Insights

Preterm ovarian hyperstimulation syndrome is a rare condition causing swelling and ovarian cysts in preterm infant girls. This case highlights spontaneous resolution of symptoms and cysts without intervention.

Area of Science:

  • Pediatric Endocrinology
  • Neonatology
  • Reproductive Endocrinology

Background:

  • Preterm ovarian hyperstimulation syndrome (POHS) is a rare condition affecting female infants born prematurely.
  • It is characterized by ovarian enlargement, follicular cysts, and elevated estradiol levels.

Observation:

  • A 23-week gestational age infant presented with elevated 17-hydroxyprogesterone, clitoromegaly, and a ventral groove at 30 weeks postconceptional age (PCA).
  • Pelvic ultrasound at 32 weeks PCA showed a normal uterus, with ovaries not visualized.
  • By 39 weeks PCA, bilateral ovarian follicular cysts were observed, measuring up to 38x25x36 mm.

Findings:

  • Hormonal levels, including estradiol and testosterone, began normalizing spontaneously by 42 weeks PCA.
  • Ovarian cysts showed regression: the right cyst resolved, and the left cyst diminished in size without treatment.

Implications:

  • This case suggests that POPS may resolve spontaneously in preterm infants.
  • It underscores the importance of serial monitoring of ovarian morphology and hormonal levels in affected neonates.
  • Further research is needed to understand the natural history and optimal management of POPS.

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