{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T10:45:03Z","timestamp":1780397103270,"version":"3.54.1"},"reference-count":15,"publisher":"BMJ","issue":"2","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2025,2,17]]},"published-print":{"date-parts":[[2025,2]]},"abstract":"<jats:p>We describe a case of an aggressive proximal humerus aneurysmal bone cyst (ABC) mimicking telangiectatic osteosarcoma with complete remodelling and reconstitution of the bone. A boy in middle childhood presented with a pathological fracture preceded by progressive right shoulder pain and swelling. Imaging and open biopsy supported the diagnosis of primary ABC with positive ubiquitin-specific protease 6 gene fusion. Treatment was initially refused, leading to significant bone erosion. Subsequently, intralesional resection and grafting were performed, confirming the ABC diagnosis. Three years post-surgery, radiographs show excellent bone remodelling, with the child maintaining premorbid shoulder function. By performing an intralesional resection, there was no doubt of tissue representation, and it allows contamination to be minimised should a wide resection be necessary later. Interestingly, there was complete reconstitution and remodelling of the proximal humerus; likely from intact periosteum like that seen in the donor sites of fibula strut grafts.<\/jats:p>","DOI":"10.1136\/bcr-2024-263437","type":"journal-article","created":{"date-parts":[[2025,2,26]],"date-time":"2025-02-26T09:20:25Z","timestamp":1740561625000},"page":"e263437","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Complete remodelling post-intralesional resection of an aggressive proximal humerus aneurysmal bone cyst mimicking telangiectatic osteosarcoma"],"prefix":"10.1136","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0009-0006-4518-7886","authenticated-orcid":false,"given":"Harpreet","family":"Singh","sequence":"first","affiliation":[{"name":"Medicine, National University of Singapore, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sze Jet","family":"Aw","sequence":"additional","affiliation":[{"name":"Anatomical Pathology, KK Women's and Children\u2019s Hospital, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Arjandas","family":"Mahadev","sequence":"additional","affiliation":[{"name":"Orthopaedic Surgery, KK Women's and Children\u2019s Hospital, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1969-7664","authenticated-orcid":false,"given":"Mohammad Ashik Bin","family":"Zainuddin","sequence":"additional","affiliation":[{"name":"Orthopaedic Surgery, KK Women's and Children\u2019s Hospital, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kenneth Pak Leung","family":"Wong","sequence":"additional","affiliation":[{"name":"Orthopaedic Surgery, KK Women's and Children\u2019s Hospital, Singapore"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2025,2,26]]},"reference":[{"key":"2025082709180554000_18.2.e263437.1","doi-asserted-by":"crossref","first-page":"81","DOI":"10.4132\/jptm.2023.02.23","article-title":"Aneurysmal bone cyst: a review","volume":"57","author":"Nasri","year":"2023","journal-title":"J Pathol Transl Med"},{"key":"2025082709180554000_18.2.e263437.2","doi-asserted-by":"publisher","DOI":"10.1007\/s12178-016-9371-6"},{"key":"2025082709180554000_18.2.e263437.3","unstructured":"Wittig JC . Telangiectatic osteosarcoma. 2022. Available: https:\/\/tumorsurgery.org\/tumor-education\/bone-tumors\/types-of-bone-tumors\/telangiectatic-osteosarcoma.aspx#:~:text=It%20is%20a%20cancerous%20tumor,cavities%20that%20fill%20with%20hemorrhage"},{"key":"2025082709180554000_18.2.e263437.4","unstructured":"Limaiem F . Telangiectatic osteosarcoma. U.S. National Library of Medicine, 2023. Available: https:\/\/www.ncbi.nlm.nih.gov\/books\/NBK537309\/"},{"key":"2025082709180554000_18.2.e263437.5","doi-asserted-by":"publisher","DOI":"10.3892\/ol.2017.5682"},{"key":"2025082709180554000_18.2.e263437.6","doi-asserted-by":"crossref","DOI":"10.1016\/j.jbo.2021.100384","article-title":"Current strategies for the treatment of solitary and aneurysmal bone cysts: A review of the literature","volume":"30","author":"Deventer","year":"2021","journal-title":"J Bone Oncol"},{"key":"2025082709180554000_18.2.e263437.7","doi-asserted-by":"crossref","DOI":"10.1503\/cmaj.200005","article-title":"Periosteum preservation in bone regeneration","volume":"192","author":"Colangeli","year":"2020","journal-title":"CMAJ"},{"key":"2025082709180554000_18.2.e263437.8","doi-asserted-by":"crossref","first-page":"2191","DOI":"10.1007\/s00264-016-3233-0","article-title":"Fibula regeneration following non-vascularized graft harvest in children","volume":"40","author":"Agarwal","year":"2016","journal-title":"Int Orthop"},{"key":"2025082709180554000_18.2.e263437.9","doi-asserted-by":"crossref","first-page":"1115","DOI":"10.1016\/j.otsr.2017.05.027","article-title":"The role of the periosteal sleeve in the reconstruction of bone defects using a non-vascularised fibula graft in the pediatric population","volume":"103","author":"Steiger","year":"2017","journal-title":"Orthop Traumatol Surg Res"},{"key":"2025082709180554000_18.2.e263437.10","doi-asserted-by":"crossref","first-page":"97","DOI":"10.1016\/S0891-8422(23)01169-2","article-title":"Bone healing in children","volume":"18","author":"Lindaman","year":"2001","journal-title":"Clin Podiatr Med Surg"},{"key":"2025082709180554000_18.2.e263437.11","doi-asserted-by":"crossref","DOI":"10.1136\/bcr-2021-245586","article-title":"Rare right acetabulum aneurysmal bone cyst treated with curettage and local adjuvants in a 4-year-old girl","volume":"14","author":"Wong","year":"2021","journal-title":"BMJ Case Rep"},{"key":"2025082709180554000_18.2.e263437.12","doi-asserted-by":"crossref","first-page":"84","DOI":"10.5005\/jp-journals-10080-1462","article-title":"Non-vascularised Fibular Autograft for Reconstruction of Paediatric Bone Defects: An Analysis of 10 Cases","volume":"15","author":"Sheridan","year":"2020","journal-title":"Strategies Trauma Limb Reconstr"},{"key":"2025082709180554000_18.2.e263437.13","doi-asserted-by":"crossref","first-page":"103108","DOI":"10.1016\/j.otsr.2021.103108","article-title":"Assessment of fibular regeneration after graft harvesting in patients with benign bone tumors: A retrospective study comparing different age groups","volume":"108","author":"Mustafa","year":"2022","journal-title":"Orthop Traumatol Surg Res"},{"key":"2025082709180554000_18.2.e263437.14","doi-asserted-by":"crossref","DOI":"10.1038\/s41598-023-29130-w","article-title":"Simple curettage and allogeneic cancellous bone chip impaction grafting in solitary enchondroma of the short tubular bones of the hand","volume":"13","author":"Park","year":"2023","journal-title":"Sci Rep"},{"key":"2025082709180554000_18.2.e263437.15","doi-asserted-by":"crossref","first-page":"e555","DOI":"10.1097\/BPO.0000000000001839","article-title":"Efficacy of Sclerotherapy With Polidocanol (Ethoxysclerol) in Primary Aneurysmal Bone Cysts in Children and Adolescents","volume":"41","author":"Jasper","year":"2021","journal-title":"J Pediatr Orthop"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2024-263437","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T16:18:15Z","timestamp":1756311495000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2024-263437"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,2]]},"references-count":15,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2025,2,26]]},"published-print":{"date-parts":[[2025,2]]}},"alternative-id":["10.1136\/bcr-2024-263437"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2024-263437","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,2]]}}}