Gorham-Stout disease--stabilization during bisphosphonate treatment.

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.

• Authors: Fabian Hammer, Werner Kenn, Ulrich Wesselmann, Lorenz C Hofbauer, Günter Delling, Bruno Allolio, Wiebke Arlt
• Journal: Journal of bone and mineral research : the official journal of the American Society for Bone and Mineral Research (J Bone Miner Res) Vol. 20 Issue 2 Pg. 350-3 (Feb 2005) ISSN: 0884-0431 [Print] United States
• PMID: 15647829 (Publication Type: Case Reports, Journal Article, Research Support, Non-U.S. Gov't)

Chemical References

• Diphosphonates
• Pamidronate

Topics

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