Gorham-Stout disease--stabilization during bisphosphonate treatment

J Bone Miner Res. 2005 Feb;20(2):350-3. doi: 10.1359/JBMR.041113. Epub 2004 Nov 16.

Abstract

A 45-year-old woman presented with recent onset of left-sided chest pain. On clinical examination, these symptoms seemed to be strictly localized to a region that was marked by a long-standing cutaneous erythematous lesion. Laboratory results showed no gross abnormalities. Radiological imaging including conventional X-ray, MRI scans, and 3D CT reconstruction of the rib cage revealed circumscript destruction of the left lateral ribs 9-11. Histological analysis of a rib biopsy showed angiomatous hypervascularization and intracortical fibrosis. In keeping with these findings, the patient's condition was diagnosed as Gorham-Stout disease, a rare condition with localized, often unilateral, bone destruction. Monotherapy with bisphosphonates (pamidronate 30 mg i.v. every 3 months) was initiated, leading to rapid disappearance of local pain. Follow-up over 24 months documented a stable clinical and radiological picture without evidence of progressive bone destruction.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Biopsy
  • Bone and Bones / blood supply*
  • Bone and Bones / pathology
  • Diphosphonates / pharmacology*
  • Diphosphonates / therapeutic use
  • Female
  • Fibrosis / pathology
  • Follow-Up Studies
  • Humans
  • Magnetic Resonance Imaging
  • Middle Aged
  • Osteolysis, Essential / diagnosis*
  • Osteolysis, Essential / drug therapy*
  • Pamidronate
  • Ribs / pathology
  • Time Factors
  • Tomography, X-Ray Computed
  • X-Rays

Substances

  • Diphosphonates
  • Pamidronate