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

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 c...

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Main Authors: Leevi A Toivonen, Marko H Neva, Thanos Sioris, Pia Isomäki, Saara Metso
Format: Article
Language:English
Published: Bioscientifica 2022-06-01
Series:Endocrinology, Diabetes & Metabolism Case Reports
Online Access:https://edm.bioscientifica.com/view/journals/edm/2022/1/EDM21-0101.xml
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author Leevi A Toivonen
Marko H Neva
Thanos Sioris
Pia Isomäki
Saara Metso
author_facet Leevi A Toivonen
Marko H Neva
Thanos Sioris
Pia Isomäki
Saara Metso
author_sort Leevi A Toivonen
collection DOAJ
description 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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spelling doaj.art-93918d4f9f044cf1804c7cf53d3436a22022-12-22T00:18:26ZengBioscientificaEndocrinology, Diabetes & Metabolism Case Reports2052-05732022-06-01111510.1530/EDM-21-0101Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case reportLeevi A Toivonen0Marko H Neva1Thanos Sioris2Pia Isomäki3Saara Metso4Department of Orthopaedic and Trauma SurgeryDepartment of Orthopaedic and Trauma SurgeryDepartment of Cardiothoracic SurgeryCentre for Rheumatic Diseases, Department of Internal MedicineUnit of Endocrinology, Department of Internal Medicine, Faculty of Medicine and Health Technology, Tampere University, Tampere University Hospital, Tampere, FinlandGorham–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.https://edm.bioscientifica.com/view/journals/edm/2022/1/EDM21-0101.xml
spellingShingle Leevi A Toivonen
Marko H Neva
Thanos Sioris
Pia Isomäki
Saara Metso
Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report
Endocrinology, Diabetes & Metabolism Case Reports
title Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report
title_full Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report
title_fullStr Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report
title_full_unstemmed Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report
title_short Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report
title_sort gorham stout disease with life threatening pleural effusion treated with a pleuro peritoneal shunt a case report
url https://edm.bioscientifica.com/view/journals/edm/2022/1/EDM21-0101.xml
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AT thanossioris gorhamstoutdiseasewithlifethreateningpleuraleffusiontreatedwithapleuroperitonealshuntacasereport
AT piaisomaki gorhamstoutdiseasewithlifethreateningpleuraleffusiontreatedwithapleuroperitonealshuntacasereport
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