Treatment of Gorham-Stout disease with zoledronic acid and interferon-α: a case report and literature review

Gorham-Stout disease is a rare disease characterized by osteolysis, angiomatosis, and soft-tissue swelling. It is a diagnosis of exclusion and has an unknown etiology. Chylothorax is a common complication of the disease that is associated with a high mortality rate. There is no standard of treatment...

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Veröffentlicht in:	Journal of pediatric hematology/oncology 2010-11, Vol.32 (8), p.579-584
Hauptverfasser:	Kuriyama, Dawn K, McElligott, Sean C, Glaser, Darryl W, Thompson, Karen S
Format:	Artikel
Sprache:	eng
Schlagworte:	Adolescent Bone Density Conservation Agents - administration & dosage Depsipeptides Diphosphonates - administration & dosage Drug Therapy, Combination Female Fusarium Humans Imidazoles - administration & dosage Interferon-alpha - administration & dosage Osteolysis, Essential - complications Osteolysis, Essential - drug therapy Osteolysis, Essential - pathology Pleural Effusion - drug therapy Pleural Effusion - etiology Pleural Effusion - pathology Polyethylene Glycols - administration & dosage Recombinant Proteins
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Zusammenfassung:	Gorham-Stout disease is a rare disease characterized by osteolysis, angiomatosis, and soft-tissue swelling. It is a diagnosis of exclusion and has an unknown etiology. Chylothorax is a common complication of the disease that is associated with a high mortality rate. There is no standard of treatment. We report a case of a 16-year-old female with Gorham-Stout disease and recurrent pleural effusions who was successfully treated with concurrent zoledronic acid and peg-interferon α-2b.
ISSN:	1077-4114 1536-3678
DOI:	10.1097/MPH.0b013e3181edb464