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Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report
SUMMARY: 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 pres...
Autores principales: | , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Bioscientifica Ltd
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9175580/ https://www.ncbi.nlm.nih.gov/pubmed/35642669 http://dx.doi.org/10.1530/EDM-21-0101 |
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author | Toivonen, Leevi A Neva, Marko H Sioris, Thanos Isomäki, Pia Metso, Saara |
author_facet | Toivonen, Leevi A Neva, Marko H Sioris, Thanos Isomäki, Pia Metso, Saara |
author_sort | Toivonen, Leevi A |
collection | PubMed |
description | SUMMARY: 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. LEARNING POINTS: Multidisciplinary approach is important in the management of rare and severe disorders such as Gorham-Stout disease. Pleuro-peritoneal shunting is a valuable option in the treatment of pleural effusion related to GSD. Interleukin-6 and bone resorption markers appear useful in measuring the disease activity of GSD. |
format | Online Article Text |
id | pubmed-9175580 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Bioscientifica Ltd |
record_format | MEDLINE/PubMed |
spelling | pubmed-91755802022-06-14 Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report Toivonen, Leevi A Neva, Marko H Sioris, Thanos Isomäki, Pia Metso, Saara Endocrinol Diabetes Metab Case Rep Novel Treatment SUMMARY: 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. LEARNING POINTS: Multidisciplinary approach is important in the management of rare and severe disorders such as Gorham-Stout disease. Pleuro-peritoneal shunting is a valuable option in the treatment of pleural effusion related to GSD. Interleukin-6 and bone resorption markers appear useful in measuring the disease activity of GSD. Bioscientifica Ltd 2022-05-03 /pmc/articles/PMC9175580/ /pubmed/35642669 http://dx.doi.org/10.1530/EDM-21-0101 Text en © The authors https://creativecommons.org/licenses/by-nc-nd/4.0/ This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. (https://creativecommons.org/licenses/by-nc-nd/4.0/) . |
spellingShingle | Novel Treatment Toivonen, Leevi A Neva, Marko H Sioris, Thanos Isomäki, Pia Metso, Saara Gorham–Stout disease with life-threatening pleural effusion treated with a pleuro-peritoneal shunt: a case report |
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 |
topic | Novel Treatment |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9175580/ https://www.ncbi.nlm.nih.gov/pubmed/35642669 http://dx.doi.org/10.1530/EDM-21-0101 |
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