A Rare Ovarian Mixed Sex Cord Stromal Tumor in a Patient with Ollier Disease: A Case Report

J Pediatr Adolesc Gynecol. 2024 Dec;37(6):629-631. doi: 10.1016/j.jpag.2024.07.009. Epub 2024 Aug 2.

Abstract

This is a case report of a 10-year-old with Ollier disease and an ovarian mass. Ollier disease, a rare disorder characterized by multiple enchondromas resulting in bone deformities, has been occasionally associated with ovarian juvenile granulosa cell tumor. This patient developed signs of precocious puberty and was found to have an ovarian tumor; however, pathology revealed a mixed sex-cord stromal tumor with components of juvenile granulosa and Sertoli-Leydig cell tumor. Tumor genomic testing revealed an IDH1 mutation. Mixed sex-cord stromal tumors of this type, also called "gynandroblastomas," have been associated with DICER1 mutations and DICER1 tumor predisposition syndrome but never with Ollier disease. Our findings expand the known spectrum of syndromic associations with this tumor type, with implications for tumor screening.

Keywords: Gynandroblastoma; Juvenile granulosa cell tumor; Mixed sex-cord stromal tumor; Ollier disease.

Publication types

  • Case Reports

MeSH terms

  • Child
  • DEAD-box RNA Helicases
  • Enchondromatosis* / complications
  • Enchondromatosis* / diagnostic imaging
  • Enchondromatosis* / genetics
  • Female
  • Granulosa Cell Tumor / complications
  • Granulosa Cell Tumor / genetics
  • Granulosa Cell Tumor / pathology
  • Humans
  • Isocitrate Dehydrogenase / genetics
  • Mutation
  • Ovarian Neoplasms* / complications
  • Ovarian Neoplasms* / genetics
  • Puberty, Precocious / etiology
  • Puberty, Precocious / genetics
  • Ribonuclease III / genetics
  • Sex Cord-Gonadal Stromal Tumors* / complications
  • Sex Cord-Gonadal Stromal Tumors* / genetics
  • Sex Cord-Gonadal Stromal Tumors* / pathology

Substances

  • Isocitrate Dehydrogenase
  • IDH1 protein, human
  • Ribonuclease III
  • DICER1 protein, human
  • DEAD-box RNA Helicases