{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,4,21]],"date-time":"2026-04-21T15:06:42Z","timestamp":1776784002901,"version":"3.51.2"},"reference-count":15,"publisher":"Ovid Technologies (Wolters Kluwer Health)","issue":"C","license":[{"start":{"date-parts":[[2024,11,1]],"date-time":"2024-11-01T00:00:00Z","timestamp":1730419200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2024,11,1]],"date-time":"2024-11-01T00:00:00Z","timestamp":1730419200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2024,10,1]],"date-time":"2024-10-01T00:00:00Z","timestamp":1727740800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["lww.com","ovid.com"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2024,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction and importance:<\/jats:title>\n                    <jats:p>Gorham syndrome, or Gorham-Stout disease, is a rare disorder characterized by spontaneous and persistent bone resorption without any known cause, leading to severe complications [2]. The clinical presentation of this condition varies widely, complicating efforts in diagnosis and treatment. In our case report, we present the story of an 88-year-old woman who bravely and gracefully faced the challenges caused by Gorham syndrome. Despite prior treatments, her condition worsened, emphasizing the need for tailored approaches.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation:<\/jats:title>\n                    <jats:p>The patient's symptoms extended to bilateral shoulders, affecting daily living. Physical examination revealed tenderness, swelling, and muscle wasting. Her blood investigation showed vitamin D deficiency and imaging confirmed complete humeral head resorption.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case discussion:<\/jats:title>\n                    <jats:p>Her case highlights the limitations of standard therapies. Variable presentations underscore the importance of thorough evaluation. Radiographic evidence revealed severe bone destruction, emphasizing the disability caused by Gorham syndrome.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion:<\/jats:title>\n                    <jats:p>Recognition of the disease as early as possible, correct diagnosis, and effective management are essential. This patient's story highlights the importance of the multi-faceted treatment approach for patients with Gorham Syndrome: medical treatment, supportive care, compassion, and tenacity to improve the quality of life.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Highlights<\/jats:title>\n                  <\/jats:sec>","DOI":"10.1016\/j.ijscr.2024.110407","type":"journal-article","created":{"date-parts":[[2024,10,5]],"date-time":"2024-10-05T10:58:26Z","timestamp":1728125906000},"update-policy":"https:\/\/doi.org\/10.1097\/lww.0000000000001000","source":"Crossref","is-referenced-by-count":1,"title":["\u201cUnraveling a rare encounter: Gorham's disease in an 88-year-old female\u201d"],"prefix":"10.1097","volume":"124","author":[{"given":"Gaurav Bir","family":"Bajracharya","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Basanta","family":"Maharjan","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Pramod","family":"Bhandari","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"276","reference":[{"issue":"6","key":"R2-89-20260421","doi-asserted-by":"crossref","first-page":"1151","DOI":"10.1016\/0002-9343(87)90959-4","article-title":"Gorham's syndrome: a case report and review of the literature","volume":"83","author":"Choma,","year":"1987","journal-title":"Am. J. Med."},{"issue":"5","key":"R4-89-20260421","doi-asserted-by":"crossref","first-page":"1136","DOI":"10.1097\/JS9.0000000000000373","article-title":"The SCARE 2023 guideline: updating consensus surgical CAse REport (SCARE) guidelines","volume":"109","author":"Sohrabi,","year":"2023","journal-title":"Int. J. Surg."},{"issue":"1","key":"R5-89-20260421","article-title":"Report of a rare case of Gorham-Stout disease of both shoulders: bisphosphonate treatment and shoulder replacement","volume":"2011","author":"Garbers,","year":"2011","journal-title":"Case Rep Rheumatol."},{"issue":"5","key":"R6-89-20260421","doi-asserted-by":"crossref","first-page":"45","DOI":"10.11648\/j.jctr.20140205.11","article-title":"Gorham's disease that gave response to radiotherapy","volume":"2","author":"Gul,","year":"2014","journal-title":"J Cancer Treat Res"},{"issue":"7","key":"R8-89-20260421","first-page":"681","article-title":"Gorham-Stout disease with multiple bone involvement\u2014challenging diagnosis of a rare disease and literature review","volume":"57","author":"Momanu,","year":"2021","journal-title":"Med"},{"issue":"3","key":"R9-89-20260421","doi-asserted-by":"crossref","first-page":"290","DOI":"10.1111\/j.1479-8077.2006.00217.x","article-title":"Intravenous pamidronate in the treatment of sternoclavicular hyperostosis","volume":"9","author":"Lu,","year":"2006","journal-title":"APLAR J. Rheumatol."},{"issue":"4","key":"R10-89-20260421","doi-asserted-by":"crossref","first-page":"296","DOI":"10.1111\/ger.12054","article-title":"Vanishing bone disease of the mandible - a case report","volume":"30","author":"Asha,","year":"2013","journal-title":"Gerodontology"},{"issue":"2","key":"R11-89-20260421","doi-asserted-by":"crossref","first-page":"157","DOI":"10.1046\/j.1365-2044.2000.055002157.x","article-title":"Gorham syndrome: anaesthetic management","volume":"55","author":"Szabo,","year":"2000","journal-title":"Anaesthesia"},{"issue":"8","key":"R12-89-20260421","doi-asserted-by":"crossref","first-page":"729","DOI":"10.1631\/jzus.B1200308","article-title":"Gorham-Stout syndrome in mainland China: a case series of 67 patients and review of the literature","volume":"14","author":"Hu,","year":"2013","journal-title":"J Zhejiang Univ Sci B"},{"issue":"9","key":"R13-89-20260421","doi-asserted-by":"crossref","first-page":"2970","DOI":"10.1002\/art.20464","article-title":"Successful treatment of SAPHO syndrome with zoledronic acid","volume":"50","author":"Kopterides,","year":"2004","journal-title":"Arthritis Rheum."},{"issue":"6","key":"R14-89-20260421","doi-asserted-by":"crossref","first-page":"613","DOI":"10.1002\/ppul.20849","article-title":"Gorham-Stout Syndrome with chylothorax: successful remission by interferon alpha-2b","volume":"44","author":"Kose,","year":"2009","journal-title":"Pediatr. Pulmonol."},{"issue":"1","key":"R15-89-20260421","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1186\/1546-0096-9-31","article-title":"The diagnosis and management of patients with idiopathic osteolysis","volume":"9","author":"Al Kaissi,","year":"2011","journal-title":"Pediatr. Rheumatol."},{"issue":"5","key":"R16-89-20260421","doi-asserted-by":"crossref","first-page":"389","DOI":"10.3415\/VCOT-11-03-0047","article-title":"Massive osteolysis in a dog resembling Gorham's disease in humans","volume":"24","author":"Farrell,","year":"2011","journal-title":"Vet. Comp. Orthop. Traumatol."},{"issue":"42","key":"R17-89-20260421","article-title":"Mandibular Gorham-Stout disease","volume":"96","author":"Liu,","year":"2017","journal-title":"Med (United States)"},{"issue":"4","key":"R18-89-20260421","doi-asserted-by":"crossref","first-page":"417","DOI":"10.1002\/ppul.22869","article-title":"Propranolol as a treatment option in Gorham-Stout syndrome: a case report","volume":"49","author":"Nir,","year":"2014","journal-title":"Pediatr. Pulmonol."}],"container-title":["International Journal of Surgery Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S221026122401188X?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S221026122401188X?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.lww.com\/10.1016\/j.ijscr.2024.110407","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,21]],"date-time":"2026-04-21T14:03:31Z","timestamp":1776780211000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.lww.com\/10.1016\/j.ijscr.2024.110407"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,11]]},"references-count":15,"journal-issue":{"issue":"C","published-print":{"date-parts":[[2024]]}},"URL":"https:\/\/doi.org\/10.1016\/j.ijscr.2024.110407","relation":{},"ISSN":["2210-2612"],"issn-type":[{"value":"2210-2612","type":"print"}],"subject":[],"published":{"date-parts":[[2024,11]]},"assertion":[{"value":"2024-08-07","name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-10-01","name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}}]}}