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Gorham-Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report

Leevi A Toivonen1, Marko H Neva1, Thanos Sioris2

  • 1Department of Orthopaedic and Trauma Surgery, Department of Internal Medicine.

Endocrinology, Diabetes & Metabolism Case Reports
|June 1, 2022
PubMed

Insights

Gorham-Stout disease (GSD) management can be improved with a multidisciplinary approach. A mini-invasive pleuro-peritoneal shunt effectively treated life-threatening pleural effusion in a GSD patient.

Area of Science:

  • Orthopedics
  • Rare Diseases
  • Oncology

Background:

  • Gorham-Stout disease (GSD) is a rare, aggressive bone disorder with unknown etiology.
  • Massive osteolysis and lymphatic proliferation characterize GSD.
  • Thoracic GSD can lead to life-threatening pleural effusion and neurological complications like paraparesis.

Purpose of the Study:

  • To report a case of thoracic Gorham-Stout disease with paraparesis and severe pleural effusion.
  • To highlight the successful management of pleural effusion using a pleuro-peritoneal shunt, a novel approach for GSD.
  • To investigate the potential role of interleukin-6 and bone resorption markers in monitoring GSD activity.

Main Methods:

  • Case report of a 23-year-old male with thoracic GSD.
  • Management involved a multidisciplinary team approach.
  • Treatment for pleural effusion utilized a mini-invasive pleuro-peritoneal shunt.

Main Results:

  • The patient experienced a good recovery following multidisciplinary management.
  • Pleuro-peritoneal shunting successfully resolved the life-threatening pleural effusion.
  • Interleukin-6 and bone resorption markers showed potential utility in assessing disease activity.

Conclusions:

  • A multidisciplinary approach is crucial for managing complex cases of Gorham-Stout disease.
  • Pleuro-peritoneal shunting offers a viable, minimally invasive option for GSD-related pleural effusion.
  • Biomarkers like IL-6 and bone resorption markers may aid in GSD activity monitoring.
Abstract

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