Difficult Therapeutic Decisions in Gorham-Stout Disease-Case Report and Review of the Literature

Int J Environ Res Public Health. 2022 Sep 16;19(18):11692. doi: 10.3390/ijerph191811692.

Abstract

Gorham-Stout disease (GSD) is a very rare, life-threatening condition characterized by the proliferation of lymphatic vessels and osteolysis. Unfortunately, no standard treatment has been determined for management of GSD. The available therapies are not equally effective and carry substantial side-effects. We report a 42-year-old female with GSD manifested in multifocal osteolysis and chronic chylothorax and ascites. The combined treatment with sirolimus and zoledronic acid due to its synergism of action was introduced. To our knowledge, this is the first Polish case report of adult patients with Gorham-Stout disease.

Keywords: lymphogenesis; osteolysis; sirolimus; therapy; zoledronic acid.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Adult
  • Female
  • Humans
  • Osteolysis*
  • Osteolysis, Essential* / diagnostic imaging
  • Osteolysis, Essential* / drug therapy
  • Sirolimus / therapeutic use
  • Zoledronic Acid / therapeutic use

Substances

  • Zoledronic Acid
  • Sirolimus

Grants and funding

This research received no external funding.