Two cases of Gorham-Stout disease with good response to zoledronic acid treatment

María Lorena Brance1,2,3, Alejandro Castiglioni4, Nicolas Cóccaro4

  • 1Centro de Reumatología, Rosario, Argentina.

Insights

Gorham-Stout disease, a rare vanishing bone syndrome, involves bone loss due to vascular proliferation. Two cases show positive responses to zoledronic acid treatment, improving pain and mobility.

Area of Science:

  • Orthopedics
  • Rheumatology
  • Pathology

Background:

  • Gorham-Stout disease (GSD), or vanishing bone syndrome, is a rare condition characterized by progressive osteolysis.
  • It results from intra-osseous proliferation of non-neoplastic vascular tissue, leading to significant bone loss.
  • GSD can present in various bones across different age groups, potentially causing severe deformities and complications.

Observation:

  • This article presents two distinct clinical cases of Gorham-Stout disease.
  • Case 1: A 56-year-old woman with a 20-year history of pain and swelling, initially misdiagnosed with SLE, eventually diagnosed with GSD.
  • Case 2: A 70-year-old man with shoulder pain and limited motion, later experiencing a pathological fracture, leading to a GSD diagnosis.

Findings:

  • Both patients received intravenous zoledronic acid (5 mg).
  • Case 1 showed progressive improvement in pain, mobility, and daily activities after the third infusion.
  • Case 2 demonstrated a good response to zoledronic acid after the first infusion.

Implications:

  • These cases suggest that zoledronic acid may be an effective treatment for Gorham-Stout disease.
  • Further research into GSD and its management is warranted given its rarity and potential severity.
  • Early diagnosis and intervention with appropriate therapies like zoledronic acid can improve patient outcomes in GSD.

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