Isolated skeletal Langerhans cell histiocytosis in adults: A case report.

Autor:	Putro YAP; Department of Orthopedics and Traumatology, RSUP Dr. Sardjito Hospital, Jl. Kesehatan Sendowo No.1, Sleman 55281, D.I.Yogyakarta, Indonesia; Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia. Electronic address: yuniarthaprabowoputro@ugm.ac.id., Magetsari R; Department of Orthopedics and Traumatology, RSUP Dr. Sardjito Hospital, Jl. Kesehatan Sendowo No.1, Sleman 55281, D.I.Yogyakarta, Indonesia; Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia., Noorrafiqi MI; Department of Orthopedics and Traumatology, RSUP Dr. Sardjito Hospital, Jl. Kesehatan Sendowo No.1, Sleman 55281, D.I.Yogyakarta, Indonesia; Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia., Dwianingsih EK; Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia; Department of Anatomical Pathology, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Dr Sardjito General Hospital, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia. Electronic address: ery_malueka@ugm.ac.id., Ekaputra E; Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia; Department of Radiology, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia., Pribadi AW; Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia; Department of Radiology, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada, Jl. Farmako, Sendowo, Sekip Utara, Sleman 55281, D.I.Yogyakarta, Indonesia. Electronic address: amri.pribadi@mail.ugm.ac.id.
Jazyk:	angličtina
Zdroj:	International journal of surgery case reports [Int J Surg Case Rep] 2024 Jul; Vol. 120, pp. 109801. Date of Electronic Publication: 2024 May 25.
DOI:	10.1016/j.ijscr.2024.109801
Abstrakt:	Introduction and Importance: LCH in adults is rarely encountered, with the preference in children and axial skeleton as predilection site. Limited understanding of adult LCH causes frequent misdiagnosis, as our experience in an adult case of LCH threw off our differential diagnosis. Case Presentation: A 21-year-old male was referred to our hospital due to pain in his right shoulder. Plain radiograph and MRI showed a solitary well-marginated lytic lesion on the distal third of the clavicle. Together with a clear history and physical exam, the benign bone cyst was suspected and we performed an open biopsy simultaneously with curettage followed by internal fixation using a bone graft. Pathology and immunohistochemistry dismissed our suspicion and confirmed LCH as the main diagnosis. At six months post-surgery, no signs of recurrence were seen on the fixated site nor complained by the patient. Discussion: Diagnosing LCH involves considering imaging appearances and patient demographics as initial clues. However, confirming the diagnosis requires a biopsy with proven CD1 expression. Currently, the majority of studies recommend confirming the diagnosis before initiating therapy. This precaution is necessary due to the unclear pathophysiology of LCH, which complicates the implementation of specific therapies. Based on benign features of skeletal lesions found from imaging, invasive treatment before biopsy confirmation still gave a satisfactory outcome despite not being in line with the current recommendation. Conclusion: Excisional biopsy and curettage in solitary LCH yield satisfactory outcomes. However, further studies are needed with larger sample sizes and interventional designs. Competing Interests: Conflict of interest statement All authors declare that there is no conflict of interest regarding the publication of this report. (Copyright © 2024 The Authors. Published by Elsevier Ltd.. All rights reserved.)
Databáze:	MEDLINE
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