Gorham Stout disease - stabilization during bisphosphonate treatment

Fabian Hammer, U Wesselmann, W Kenn, LC Hofbauer, G Delling, B Allolio, Wiebke Arlt

    Research output: Contribution to journalArticle

    73 Citations (Scopus)

    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.
    Original languageEnglish
    Pages (from-to)350-353
    Number of pages4
    JournalJournal of Bone and Mineral Research
    Volume20
    DOIs
    Publication statusPublished - 1 Feb 2005

    Keywords

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

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