{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T08:10:19Z","timestamp":1756282219906,"version":"3.44.0"},"reference-count":14,"publisher":"BMJ","issue":"11","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2022,11,14]]},"published-print":{"date-parts":[[2022,11]]},"abstract":"<jats:p>Gorham-Stout disease (GSD) is thought to be due to uncontrolled proliferation of vascular and lymphatic structures within bone tissue causing destruction and osteolysis of bone. We present a patient in her mid-40s who reported chronic shoulder pain, a pleural effusion and irregular periods. Investigations showed osteolysis of her ribs, pleural effusions, an ovarian mass and a raised carbohydrate antigen 125 (Ca-125). She was subsequently diagnosed with GSD, and referred to gynaecology-oncology in consideration of potential ovarian malignancy. GSD is a diagnosis of exclusion that requires a high degree of clinical suspicion, as well as multiple investigations to achieve diagnosis. Clinicians rely on a small number of case reports to provide guidance for this. Therefore, this report provides an overview of a rare pathology, considers the differentials of a raised Ca-125 and describes how a pleural effusion, which links them both, alarmed us regarding an incidental finding of an ovarian cyst.<\/jats:p>","DOI":"10.1136\/bcr-2022-250477","type":"journal-article","created":{"date-parts":[[2022,11,22]],"date-time":"2022-11-22T10:40:55Z","timestamp":1669113655000},"page":"e250477","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":2,"title":["Thoracic Gorham-Stout disease masquerading as an ovarian malignancy: a differential diagnosis in non-malignant causes of raised Ca-125 and overview of this rare pathology"],"prefix":"10.1136","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7216-3560","authenticated-orcid":false,"given":"Alisha","family":"Sattar","sequence":"first","affiliation":[{"name":"Department of Gynaecology Oncology, Oxford University Hospitals NHS Foundation Trust, Oxford, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7407-8247","authenticated-orcid":false,"given":"Sarah","family":"Smyth","sequence":"additional","affiliation":[{"name":"Department of Gynaecology Oncology, Oxford University Hospitals NHS Foundation Trust, Oxford, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3293-5321","authenticated-orcid":false,"given":"Hooman","family":"Soleymani majd","sequence":"additional","affiliation":[{"name":"Department of Gynaecology Oncology, Oxford University Hospitals NHS Foundation Trust, Oxford, UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2022,11,22]]},"reference":[{"key":"2025082700292063000_15.11.e250477.1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM183807110182305"},{"key":"2025082700292063000_15.11.e250477.2","first-page":"302","article-title":"Hemangiomatosis and its relation to massive osteolysis","volume":"67","author":"Gorham","year":"1954","journal-title":"Trans Assoc Am Physicians"},{"key":"2025082700292063000_15.11.e250477.3","doi-asserted-by":"publisher","DOI":"10.1302\/0301-620X.81B3.9468"},{"key":"2025082700292063000_15.11.e250477.4","doi-asserted-by":"crossref","first-page":"e467","DOI":"10.1016\/j.spinee.2016.02.020","article-title":"Surgical management of Gorham disease involving the upper cervical spine with occipito-cervical-thoracic fusion: a case report","volume":"16","author":"Schell","year":"2016","journal-title":"Spine J"},{"key":"2025082700292063000_15.11.e250477.5","doi-asserted-by":"crossref","first-page":"826","DOI":"10.3346\/jkms.2002.17.6.826","article-title":"Chylothorax in Gorham's disease","volume":"17","author":"Lee","year":"2002","journal-title":"J Korean Med Sci"},{"key":"2025082700292063000_15.11.e250477.6","doi-asserted-by":"publisher","DOI":"10.1016\/0030-4220(83)90185-8"},{"key":"2025082700292063000_15.11.e250477.7","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2014.02.011"},{"key":"2025082700292063000_15.11.e250477.8","doi-asserted-by":"crossref","first-page":"401","DOI":"10.1002\/pbc.24832","article-title":"Novel molecular pathways in Gorham disease: implications for treatment","volume":"61","author":"Hagendoorn","year":"2014","journal-title":"Pediatr Blood Cancer"},{"key":"2025082700292063000_15.11.e250477.9","doi-asserted-by":"crossref","first-page":"813","DOI":"10.1007\/s10067-014-2780-2","article-title":"Gorham-Stout disease: radiological, histological, and clinical features of 12 cases and review of literature","volume":"35","author":"Liu","year":"2016","journal-title":"Clin Rheumatol"},{"key":"2025082700292063000_15.11.e250477.10","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":"2025082700292063000_15.11.e250477.11","doi-asserted-by":"crossref","unstructured":"Scatena R . [Advances in Experimental Medicine and Biology] Advances in Cancer Biomarkers Volume 867 || The Role of CA 125 as Tumor Marker: Biochemical and Clinical Aspects 2015;14:229\u201344.doi:10.1007\/978-94-017-7215-0","DOI":"10.1007\/978-94-017-7215-0_14"},{"key":"2025082700292063000_15.11.e250477.12","doi-asserted-by":"crossref","first-page":"339","DOI":"10.1016\/j.chest.2017.05.018","article-title":"CA-125 in disease progression and treatment of lymphangioleiomyomatosis","volume":"153","author":"Glasgow","year":"2018","journal-title":"Chest"},{"key":"2025082700292063000_15.11.e250477.13","first-page":"21","article-title":"Raised CA125 \u2013 what we actually know\u2026","volume":"23","author":"Howe","year":"2021","journal-title":"Obstet Gynecol"},{"key":"2025082700292063000_15.11.e250477.14","unstructured":"Royal College of Obstetricians and Gynaecologists . Management of Suspected Ovarian Masses in Premenopausal Women. [Internet] RCOG\/ BSGE Joint Guideline, 2011. Available: https:\/\/www.rcog.org.uk\/globalassets\/documents\/guidelines\/gtg_62.pdf"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2022-250477","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T07:29:30Z","timestamp":1756279770000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2022-250477"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,11]]},"references-count":14,"journal-issue":{"issue":"11","published-online":{"date-parts":[[2022,11,22]]},"published-print":{"date-parts":[[2022,11]]}},"alternative-id":["10.1136\/bcr-2022-250477"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2022-250477","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2022,11]]}}}