Endocrinology, Diabetes & Metabolism Case Reports (Jun 2022)

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

  • Leevi A Toivonen,
  • Marko H Neva,
  • Thanos Sioris,
  • Pia Isomäki,
  • Saara Metso

DOI
https://doi.org/10.1530/EDM-21-0101
Journal volume & issue
Vol. 1, no. 1
pp. 1 – 5

Abstract

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Gorham–Stout disease (GSD) is a rare bone disease characterized by massive osteolysis and lymphatic proliferation. The origin of the condition is unknown, and no established treatment protocol exists. Massive pleural effusion is a frequent complication of GSD in the thoracic region. We present the case of a 23-year-old male with thoracic GSD, subsequent paraparesis, and life-threatening pleural effusion. The patient was managed by a multidisciplinary team with a good recovery. The pleural effusion was successfully treated with a pleuro-peritoneal shunt. This is the first report of the use of this mini-invasive technique in the management of pleural effusion related to GSD. Further, we present the potential role of interleukin-6 and bone resorption markers in the measurement of the disease activity.