4.6 Article

Gorham-Stout disease - Stabilization during bisphosphonate treatment

期刊

JOURNAL OF BONE AND MINERAL RESEARCH
卷 20, 期 2, 页码 350-353

出版社

WILEY
DOI: 10.1359/JBMR.041113

关键词

Gorham-Stout disease; vanishing bone disease; local osteolysis; pamidronate; interleukin-6; osteoprotegerin; RANKL

资金

  1. Medical Research Council [G116/172] Funding Source: Medline
  2. MRC [G116/172] Funding Source: UKRI
  3. Medical Research Council [G116/172] Funding Source: researchfish

向作者/读者索取更多资源

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 hypervas-cularization 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 IV 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.

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