{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,11,29]],"date-time":"2025-11-29T23:29:42Z","timestamp":1764458982235,"version":"3.44.0"},"reference-count":18,"publisher":"BMJ","issue":"6","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2022,5,31]]},"published-print":{"date-parts":[[2022,6]]},"abstract":"<jats:p>Rhabdomyosarcoma is the most common soft-tissue sarcoma in paediatric patients and may arise as a primary orbital neoplasm. Imaging studies show a unilateral solitary orbital lesion. With larger tumours, erosion of the adjacent orbital walls is a common accompanying bony change. We present an unusual case of rhabdomyosarcoma in a preschool girl with proptosis and temporal bossing. Imaging studies showed a homogeneous, well-delineated, extraconal mass of the right orbit with hyperostosis of the adjacent sphenoid bone. After excision of the mass and debulking of the sphenoid bone, histopathology showed embryonal rhabdomyosarcoma with no malignant infiltration of the adjacent bone. This is the first reported case of hyperostosis associated with rhabdomyosarcoma. Periosteal reaction is the proposed underlying mechanism.<\/jats:p>","DOI":"10.1136\/bcr-2021-245466","type":"journal-article","created":{"date-parts":[[2022,6,20]],"date-time":"2022-06-20T12:50:54Z","timestamp":1655729454000},"page":"e245466","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":3,"title":["Hyperostosis in orbital rhabdomyosarcoma"],"prefix":"10.1136","volume":"15","author":[{"given":"Paolo Nico Aseron","family":"Rogelio","sequence":"first","affiliation":[{"name":"Department of Ophthalmology and Visual Sciences, Philippine General Hospital, Manila, Philippines"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Felice Katrina Trio","family":"Ranche","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology and Visual Sciences, Philippine General Hospital, Manila, Philippines"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Mary Rose","family":"Pe-Yan","sequence":"additional","affiliation":[{"name":"Department of Ophthalmology and Visual Sciences, Philippine General Hospital, Manila, Philippines"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2022,6,20]]},"reference":[{"key":"2025082703323715000_15.6.e245466.1","unstructured":"Wyant T , Alteri R , Kalidas M . Rhabdomyosarcoma. American Cancer Society. Available: https:\/\/www.cancer.org\/content\/dam\/CRC\/PDF\/Public\/8803.00.pdf [Accessed Jul 2021]."},{"key":"2025082703323715000_15.6.e245466.2","doi-asserted-by":"publisher","DOI":"10.3174\/ajnr.A4676"},{"key":"2025082703323715000_15.6.e245466.3","article-title":"Etiologies of proptosis: a review","volume":"6","author":"Topilow","year":"2020","journal-title":"Intern Med Rev"},{"key":"2025082703323715000_15.6.e245466.4","first-page":"444","article-title":"Myeloid sarcoma in children - diagnostic and therapeutic difficulties","volume":"20","author":"Samborska","year":"2016","journal-title":"Contemp Oncol"},{"key":"2025082703323715000_15.6.e245466.5","doi-asserted-by":"crossref","DOI":"10.1259\/bjr.20160710","article-title":"Unravelling chloroma: review of imaging findings","volume":"90","author":"Singh","year":"2017","journal-title":"Br J Radiol"},{"key":"2025082703323715000_15.6.e245466.6","doi-asserted-by":"publisher","DOI":"10.1542\/pir.2017-0087"},{"key":"2025082703323715000_15.6.e245466.7","doi-asserted-by":"publisher","DOI":"10.1038\/sj.eye.6701912"},{"key":"2025082703323715000_15.6.e245466.8","doi-asserted-by":"crossref","DOI":"10.1097\/MD.0000000000017038","article-title":"Orbital neuroblastoma metastasis: a case report and literature review","volume":"98","author":"Yang","year":"2019","journal-title":"Medicine"},{"key":"2025082703323715000_15.6.e245466.9","doi-asserted-by":"crossref","first-page":"174","DOI":"10.1016\/j.jpeds.2020.10.056","article-title":"Langerhans cell histiocytosis of the orbit: spectrum of clinical and imaging findings","volume":"230","author":"Lakatos","year":"2021","journal-title":"J Pediatr"},{"key":"2025082703323715000_15.6.e245466.10","doi-asserted-by":"crossref","first-page":"1035","DOI":"10.1016\/j.jaad.2017.05.059","article-title":"Langerhans cell histiocytosis in children: history, classification, pathobiology, clinical manifestations, and prognosis","volume":"78","author":"Krooks","year":"2018","journal-title":"J Am Acad Dermatol"},{"key":"2025082703323715000_15.6.e245466.11","doi-asserted-by":"crossref","first-page":"e679","DOI":"10.1016\/j.wneu.2019.02.123","article-title":"Spheno-Orbital meningioma: surgical outcomes and management of recurrence","volume":"126","author":"Nagahama","year":"2019","journal-title":"World Neurosurg"},{"key":"2025082703323715000_15.6.e245466.12","doi-asserted-by":"crossref","first-page":"35","DOI":"10.4103\/jpn.JPN_56_20","article-title":"Pediatric skull base tumors: a management challenge","volume":"16","author":"Ballestero","year":"2021","journal-title":"J Pediatr Neurosci"},{"key":"2025082703323715000_15.6.e245466.13","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.24118"},{"key":"2025082703323715000_15.6.e245466.14","doi-asserted-by":"crossref","unstructured":"Ermoian RP , Breneman J , Walterhouse DO . 45 Gy is not sufficient radiotherapy dose for Group III orbital embryonal rhabdomyosarcoma after less than complete response to 12 weeks of ARST0331 chemotherapy: A report from the Soft Tissue Sarcoma Committee of the Children\u2019s Oncology Group. Pediatr Blood Cancer 2017;64.","DOI":"10.1002\/pbc.26540"},{"key":"2025082703323715000_15.6.e245466.15","doi-asserted-by":"publisher","DOI":"10.1148\/rg.276075138"},{"key":"2025082703323715000_15.6.e245466.16","doi-asserted-by":"publisher","DOI":"10.1016\/j.crad.2004.08.017"},{"key":"2025082703323715000_15.6.e245466.17","first-page":"18","article-title":"Hyperostosis - an unusual radiographic presentation of Myelodysplastic Syndrome transformed to Acute Myeloid Leukemia","volume":"4","author":"Franco","year":"2010","journal-title":"J Radiol Case Rep"},{"key":"2025082703323715000_15.6.e245466.18","first-page":"16","article-title":"The accuracy of radiological imaging in the diagnosis of orbital lesions in a regional hospital","volume":"9","author":"AC","year":"2005","journal-title":"Hong Kong J Ophthalmol"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2021-245466","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,27]],"date-time":"2025-08-27T10:32:42Z","timestamp":1756290762000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2021-245466"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,6]]},"references-count":18,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2022,6,20]]},"published-print":{"date-parts":[[2022,6]]}},"alternative-id":["10.1136\/bcr-2021-245466"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2021-245466","relation":{},"ISSN":["1757-790X"],"issn-type":[{"type":"electronic","value":"1757-790X"}],"subject":[],"published":{"date-parts":[[2022,6]]}}}