McCune-Albright syndrome (MAS): early and extensive bone fibrous dysplasia involvement and "mistaken identity" oophorectomy

J Pediatr Endocrinol Metab. 2010 Aug;23(8):837-42. doi: 10.1515/jpem.2010.135.

Abstract

Background: McCune-Albright syndrome (MAS) is a triad of gonadotropin-independent precocious puberty (GIPP), café-au-lait spots (CALS) and fibrous dysplasia (FD) of bone. The extent of the abnormalities is variable.

Patient and results: We report a 3 year old girl with CALS since infancy, FD diagnosed at age of 2.5 years, and at the age of 3 years vaginal bleeding. The ultrasound revealed a cystic mass of the ovary, surgical pathology found ovarian cyst. LHRH stimulation demonstrated GIPP (LH 9.8 mIU/ml and FSH 8.9 mIU/ml; normal LH 1.8-10, FSH 9-26 mIU/ml). Radiographs and bone scans demonstrated FD in multiple bones. Peripheral leucocytes and the ovary were negative for GNAS gene mutations. Treatment with Letrasole interrupted the pubertal development.

Conclusions: We conclude that the clinical signs of MAS are telling and that timely MAS diagnosis prevents unnecessary oophorectomy. A close follow up is recommended regarding development of endocrine disorders and spreading of FD.

Publication types

  • Case Reports

MeSH terms

  • Aromatase Inhibitors / therapeutic use
  • Child, Preschool
  • Female
  • Fibrous Dysplasia, Polyostotic / diagnosis*
  • Fibrous Dysplasia, Polyostotic / drug therapy
  • Follicle Stimulating Hormone / blood
  • Gonadotropin-Releasing Hormone / blood
  • Humans
  • Letrozole
  • Luteinizing Hormone / blood
  • Nitriles / therapeutic use
  • Ovarian Cysts / diagnostic imaging
  • Ovarian Cysts / surgery
  • Ovariectomy
  • Puberty, Precocious / diagnosis
  • Triazoles / therapeutic use
  • Ultrasonography
  • Uterine Hemorrhage / pathology

Substances

  • Aromatase Inhibitors
  • Nitriles
  • Triazoles
  • Gonadotropin-Releasing Hormone
  • Letrozole
  • Luteinizing Hormone
  • Follicle Stimulating Hormone