{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,3]],"date-time":"2026-07-03T00:23:28Z","timestamp":1783038208882,"version":"3.54.6"},"reference-count":9,"publisher":"BMJ","issue":"1","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2020,12,2]]},"published-print":{"date-parts":[[2021,1]]},"abstract":"<jats:p>Gorham-Stout disease (GSD) is an extremely rare musculoskeletal disease of unknown aetiology characterised by non-neoplastic proliferation of vascular and lymphatic channels causing massive osteolysis, typically affecting younger individuals. Chylothorax is a known complication of GSD which is postulated to occur from thoracic spine involvement leading to pleural or thoracic duct invasion. In our case, bilateral chylothorax developed in a 60-year-old woman without any thoracic spine involvement of her disease, challenging the proposed mechanism. Despite bilateral pleural drainage and escalating doses of sirolimus, she ultimately developed respiratory failure and shock and succumbed to her illness. Overall survival of GSD is unknown, but when complicated by chylothorax, prognosis is typically poor. GSD represents a diagnostic and management challenge due to the paucity of knowledge surrounding its aetiology and management. These patients require multidisciplinary coordinated care. It is also important to note its high mortality when associated with chylothorax in particular.<\/jats:p>","DOI":"10.1136\/bcr-2020-237638","type":"journal-article","created":{"date-parts":[[2021,1,19]],"date-time":"2021-01-19T17:05:57Z","timestamp":1611075957000},"page":"e237638","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":10,"title":["Gorham-Stout disease of the mandible, manubrium and cervical spine presenting as bilateral chylothorax"],"prefix":"10.1136","volume":"14","author":[{"given":"Ashley Ann","family":"Thompson","sequence":"first","affiliation":[{"name":"Internal Medicine, University of Rochester Medical Center, Rochester, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara","family":"Patrawala","sequence":"additional","affiliation":[{"name":"Internal Medicine, University of Rochester Medical Center, Rochester, New York, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2021,1,19]]},"reference":[{"key":"2025082804063859000_14.1.e237638.1","doi-asserted-by":"crossref","first-page":"44","DOI":"10.5090\/kjtcs.2019.52.1.44","article-title":"Chylous manifestations and management of Gorham-Stout syndrome","volume":"52","author":"Cho","year":"2019","journal-title":"Korean J Thorac Cardiovasc Surg"},{"key":"2025082804063859000_14.1.e237638.2","doi-asserted-by":"crossref","first-page":"195","DOI":"10.1016\/j.pulmoe.2019.01.008","article-title":"Chylothorax as a debut form of Gorham-Stout disease","volume":"25","author":"Ferreiro","year":"2019","journal-title":"Pulmonology"},{"key":"2025082804063859000_14.1.e237638.3","doi-asserted-by":"publisher","DOI":"10.1097\/01.mph.0000203721.83566.e6"},{"key":"2025082804063859000_14.1.e237638.4","doi-asserted-by":"crossref","first-page":"57","DOI":"10.1016\/j.clon.2004.09.015","article-title":"Gorham's syndrome","volume":"17","author":"Gillham","year":"2005","journal-title":"Clin Oncol"},{"key":"2025082804063859000_14.1.e237638.5","doi-asserted-by":"crossref","first-page":"618","DOI":"10.1590\/S0102-36162010000600019","article-title":"Gorham-Stout syndrome: phantom bone disease","volume":"45","author":"El-Kouba","year":"2010","journal-title":"Rev Bras Ortop"},{"key":"2025082804063859000_14.1.e237638.6","doi-asserted-by":"crossref","DOI":"10.1186\/s12891-019-2542-z","article-title":"A rare case of Gorham-stout syndrome involving the thoracic spine with progressive bilateral chylothorax: a case report","volume":"20","author":"Wang","year":"2019","journal-title":"BMC Musculoskelet Disord"},{"key":"2025082804063859000_14.1.e237638.7","doi-asserted-by":"publisher","DOI":"10.1378\/chest.105.1.208"},{"key":"2025082804063859000_14.1.e237638.8","doi-asserted-by":"crossref","DOI":"10.1186\/s12891-020-03540-7","article-title":"Gorham-Stout disease successfully treated with sirolimus (rapamycin): a case report and review of the literature","volume":"21","author":"Liang","year":"2020","journal-title":"BMC Musculoskelet Disord"},{"key":"2025082804063859000_14.1.e237638.9","doi-asserted-by":"publisher","DOI":"10.1097\/MPH.0000000000000514"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2020-237638","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,28]],"date-time":"2025-08-28T11:06:48Z","timestamp":1756379208000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2020-237638"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,1]]},"references-count":9,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2021,1,19]]},"published-print":{"date-parts":[[2021,1]]}},"alternative-id":["10.1136\/bcr-2020-237638"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2020-237638","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,1]]}}}