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:10.1530/EDM-21-0101

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

TAYS Heart Centre

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Abstract

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.

Original language	English
Article number	21-0101
Number of pages	5
Journal	Endocrinology, Diabetes and Metabolism Case Reports
Volume	2022
Issue number	1
DOIs	https://doi.org/10.1530/EDM-21-0101
Publication status	Published - 2022
Publication type	A1 Journal article-refereed

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Internal Medicine
Endocrinology, Diabetes and Metabolism

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[20520573 - Endocrinology, Diabetes & Metabolism Case Reports] Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt a case report-1Final published version, 1.04 MBLicence: CC BY-NC-ND

https://urn.fi/URN:NBN:fi:tuni-202209096966Licence: CC BY-NC-ND

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

abstract = "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.",

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T1 - Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt

T2 - a case report

AU - Toivonen, Leevi A.

AU - Neva, Marko H.

AU - Sioris, Thanos

AU - Isomäki, Pia

AU - Metso, Saara

PY - 2022

Y1 - 2022

N2 - 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.

AB - 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.

U2 - 10.1530/EDM-21-0101

DO - 10.1530/EDM-21-0101

M3 - Article

AN - SCOPUS:85133415517

SN - 2052-0573

VL - 2022

JO - Endocrinology, Diabetes and Metabolism Case Reports

JF - Endocrinology, Diabetes and Metabolism Case Reports

IS - 1

M1 - 21-0101

ER -

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

Abstract

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