{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T01:12:49Z","timestamp":1790903569205,"version":"4.1.0"},"reference-count":21,"publisher":"MDPI AG","issue":"2","license":[{"start":{"date-parts":[[2022,2,3]],"date-time":"2022-02-03T00:00:00Z","timestamp":1643846400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"National Research Foundation of Korea (NRF) grant funded by the Government of South Korea (Ministry of Science and ICT, MSIT)","award":["NRF-2020R1C1C1006623"],"award-info":[{"award-number":["NRF-2020R1C1C1006623"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Diagnostics"],"abstract":"<jats:p>Langerhans cell histiocytosis (LCH) is a rare neoplastic disorder characterized by the clonal proliferation of CD1a +\/CD 207 + dendritic cells, whose features are similar to those of epidermal Langerhans cells. LCH is more common in children than in adults. Localized osteolytic lesions in the craniofacial bones are the most common manifestations of LCH. However, LCH can also present as a multifocal and multisystem disease with poor prognosis. Locally aggressive LCH needs to be differentiated from various diseases such as osteomyelitis, malignant bone tumors, and soft tissue sarcomas. However, it is difficult to diagnose, since the imaging findings are nonspecific. We report a case of a highly aggressive LCH in the maxilla accompanied by a fluid-fluid level.<\/jats:p>","DOI":"10.3390\/diagnostics12020400","type":"journal-article","created":{"date-parts":[[2022,2,4]],"date-time":"2022-02-04T11:35:17Z","timestamp":1643974517000},"page":"400","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Diagnostic Challenge in Rapidly Growing Langerhans Cell Histiocytosis with Aneurysmal Bone Cyst in the Maxilla: A Case Report"],"prefix":"10.3390","volume":"12","author":[{"given":"Ye Rin","family":"Hwang","sequence":"first","affiliation":[{"name":"Department of Radiology, Kyung Hee University College of Medicine, Kyung Hee University Hospital, #23 Kyunghee-daero, Dongdaemun-gu, Seoul 02447, Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3424-0208","authenticated-orcid":false,"given":"Kyung Mi","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Radiology, Kyung Hee University College of Medicine, Kyung Hee University Hospital, #23 Kyunghee-daero, Dongdaemun-gu, Seoul 02447, Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hyug-Gi","family":"Kim","sequence":"additional","affiliation":[{"name":"Department of Radiology, Kyung Hee University College of Medicine, Kyung Hee University Hospital, #23 Kyunghee-daero, Dongdaemun-gu, Seoul 02447, Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4220-6755","authenticated-orcid":false,"given":"Kiyong","family":"Na","sequence":"additional","affiliation":[{"name":"Department of Pathology, Kyung Hee University College of Medicine, Kyung Hee University Hospital, #23 Kyunghee-daero, Dongdaemun-gu, Seoul 02447, Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2022,2,3]]},"reference":[{"key":"ref_1","first-page":"2008","article-title":"More than just Langerhans cell histiocytosis: A radiologic review of histiocytic disorders","volume":"34","author":"Zaveri","year":"2014","journal-title":"Radiogr. Rev. Publ. Radiol. Soc. N. Am. Inc."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"569","DOI":"10.1007\/s13244-013-0271-7","article-title":"Skeletal involvement in Langerhans cell histiocytosis","volume":"4","author":"Khung","year":"2013","journal-title":"Insights Into Imaging"},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"327","DOI":"10.1007\/s00234-017-1798-y","article-title":"Non-odontogenic tumors of the facial bones in children and adolescents: Role of multiparametric imaging","volume":"59","author":"Becker","year":"2017","journal-title":"Neuroradiology"},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"1874","DOI":"10.1016\/j.ijporl.2014.08.015","article-title":"Head and neck manifestations of langerhan\u2019s cell histiocytosis in children: A 46-year experience","volume":"78","author":"Lewoczko","year":"2014","journal-title":"Int. J. Pediatric Otorhinolaryngol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"97","DOI":"10.1016\/j.ejro.2019.02.002","article-title":"Secondary aneurysmal bone cyst in Langerhans cell histiocytosis: Case report, literature review","volume":"6","author":"Lomoro","year":"2019","journal-title":"Eur. J. Radiol. Open"},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"589","DOI":"10.2214\/AJR.07.3573","article-title":"Craniofacial and intracranial manifestations of langerhans cell histiocytosis: Report of findings in 100 patients","volume":"191","author":"Soohoo","year":"2008","journal-title":"AJR Am. J. Roentgenol."},{"key":"ref_7","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. Derm."},{"key":"ref_8","first-page":"5","article-title":"Langerhans Cell Histiocytosis with Multiple Fluid-fluid Levels in the Parietal Bone","volume":"19","author":"Matsushita","year":"2020","journal-title":"Magn. Reson. Med. Sci. MRMS Off. J. Jpn. Soc. Magn. Reson. Med."},{"key":"ref_9","first-page":"1258","article-title":"Orbital histiocytosis X","volume":"16","author":"Erly","year":"1995","journal-title":"AJNR Am. J. Neuroradiol."},{"key":"ref_10","first-page":"964","article-title":"Langerhans cell histiocytosis involving the sphenoid sinus and superior orbital fissure","volume":"16","author":"Stromberg","year":"1995","journal-title":"AJNR Am. J. Neuroradiol."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"814","DOI":"10.1016\/S0002-9394(14)71699-X","article-title":"Langerhans cell histiocytosis with orbital involvement","volume":"124","author":"Kramer","year":"1997","journal-title":"Am. J. Ophthalmol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"e92","DOI":"10.5114\/pjr.2019.83101","article-title":"A systematic approach in the diagnosis of paediatric skull lesions: What radiologists need to know","volume":"84","author":"Choudhary","year":"2019","journal-title":"Pol. J. Radiol."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"157","DOI":"10.1148\/rg.261055050","article-title":"Causes of facial swelling in pediatric patients: Correlation of clinical and radiologic findings","volume":"26","author":"Khanna","year":"2006","journal-title":"Radiographics"},{"key":"ref_14","first-page":"47","article-title":"Fluid-fluid level in Langerhans cell histiocytosis of the skull","volume":"11","author":"Albadr","year":"2006","journal-title":"Neurosciences"},{"key":"ref_15","first-page":"20150408","article-title":"Langerhans cell histiocytosis: Another cause of a fluid-fluid level within an appendicular bony lesion","volume":"2","author":"Leong","year":"2016","journal-title":"BJR Case Rep."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"604","DOI":"10.3174\/ajnr.A3712","article-title":"CT and MRI of pediatric skull lesions with fluid-fluid levels","volume":"35","author":"Nabavizadeh","year":"2014","journal-title":"AJNR Am. J. Neuroradiol."},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"949","DOI":"10.1038\/nature00766","article-title":"Mutations of the BRAF gene in human cancer","volume":"417","author":"Davies","year":"2002","journal-title":"Nature"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"272","DOI":"10.1186\/s13023-021-01912-3","article-title":"Frequency detection of BRAF V600E mutation in a cohort of pediatric langerhans cell histiocytosis patients by next-generation sequencing","volume":"16","author":"Feng","year":"2021","journal-title":"Orphanet J. Rare Dis."},{"key":"ref_19","first-page":"122","article-title":"Pediatric Langerhans cell histiocytosis: State of the science and future directions","volume":"17","author":"Thacker","year":"2019","journal-title":"Clin. Adv. Hematol. Oncol"},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"669","DOI":"10.1084\/jem.20130977","article-title":"BRAF-V600E expression in precursor versus differentiated dendritic cells defines clinically distinct LCH risk groups","volume":"211","author":"Berres","year":"2014","journal-title":"J. Exp. Med."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"319","DOI":"10.1084\/jem.20161881","article-title":"RAF\/MEK\/extracellular signal-related kinase pathway suppresses dendritic cell migration and traps dendritic cells in Langerhans cell histiocytosis lesions","volume":"215","author":"Hogstad","year":"2018","journal-title":"J. Exp. Med."}],"container-title":["Diagnostics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2075-4418\/12\/2\/400\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T22:13:50Z","timestamp":1760134430000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2075-4418\/12\/2\/400"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,2,3]]},"references-count":21,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2022,2]]}},"alternative-id":["diagnostics12020400"],"URL":"https:\/\/doi.org\/10.3390\/diagnostics12020400","relation":{},"ISSN":["2075-4418"],"issn-type":[{"value":"2075-4418","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,2,3]]}}}