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Gorham-Stout Disease—Stabilization During Bisphosphonate Treatment

2005; Oxford University Press; Volume: 20; Issue: 2 Linguagem: Inglês

10.1359/jbmr.041113

1523-4681

Fabian Hammer, W. Kenn, Ulrich Wesselmann, Lorenz C. Hofbauer, G. Delling, Bruno Allolio, Wiebke Arlt,

Oral and gingival health research

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 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.