A Child with Early-Onset Gorham-Stout Disease Complicated by Chylothorax: Near-Complete Regression of Bone Lesions with Interferon and Bisphosphonate Treatment

Horm Res Paediatr. 2019;91(6):406-410. doi: 10.1159/000495364. Epub 2019 Jan 10.

Abstract

We report a case of Gorham-Stout disease (GSD) complicated by chylothorax and treated with a combination therapy with interferon and bisphosphonates. This treatment may be helpful in improving the usually unfavorable prognosis of GSD beginning with a chylothorax before 1 year of age, and in reducing bone lesions. Moreover, the use of bisphosphonates appears to be useful in treating pain.

Keywords: Alpha-2b interferon; Bisphosphonate; Chylothorax; Gorham-Stout disease.

Publication types

  • Case Reports

MeSH terms

  • Chylothorax* / drug therapy
  • Chylothorax* / pathology
  • Chylothorax* / physiopathology
  • Diphosphonates / administration & dosage*
  • Humans
  • Infant
  • Interferon-alpha / administration & dosage*
  • Male
  • Osteolysis, Essential* / drug therapy
  • Osteolysis, Essential* / pathology
  • Osteolysis, Essential* / physiopathology
  • Pain* / drug therapy
  • Pain* / pathology
  • Pain* / physiopathology

Substances

  • Diphosphonates
  • IFNA2 protein, human
  • Interferon-alpha