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Isolated Pulmonary Langerhans Cell Histiocytosis in an Infant Diagnosed by CD1a Immunostaining of Bronchoalveolar Lavage Cells: A Case Report and Literature Review
Clin Pediatr Hematol Oncol 2017;24:157-61.
Published online October 31, 2017
© 2017 Korean Society of Pediatric Hematology-Oncology and Korean Society for Pediatric Neuro-Oncology

Mi Jin Kim, M.D.1, Jin Ho Yoo, M.D.1, Young Wook Cho, M.D.2, Jae Won Yoo, M.D.1, Sung Han Kang, M.D.1, Hyery Kim, M.D.1, Kyung Nam Koh, M.D.1, Ho Joon Im, M.D.1 and Jong Jin Seo, M.D., Ph.D.1

Departments of 1Pediatrics, 2Laboratory Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea
Correspondence to: Jong Jin Seo
Division of Pediatric Hematology/ Oncology, Department of Pediatrics, Asan Medical Center Children’s Hospital, University of Ulsan College of Medicine, 88, Olympic-ro 43-gil, Songpa-gu, Seoul 05505, Korea
Tel: +82-2-3010-3383 Fax: +82-2-473-3725 E-mail: jjseo@amc.seoul.kr
Received September 25, 2017; Revised October 8, 2017; Accepted October 16, 2017.
This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Abstract
Isolated pulmonary Langerhans cell histiocytosis (LCH) is a very rare disease in childhood. We report a case of a 5-month-old girl with isolated pulmonary LCH, who was transferred due to incidental chest x-ray finding of multiple cystic lesions without any clinical symptoms. Chest computed tomography (CT) finding suggested that pulmonary LCH was likely, but evaluations including lung biopsy were negative. At a follow-up visit three months later, we performed bronchoalveolar lavage (BAL) fluid analysis and confirmed the presence of CD1a-positive cells, thereby confirming diagnosis of pulmonary LCH. After completing eight months of chemotherapy, yearly follow-up evaluations were performed and there has been no evidence of reactivation of the disease for four years. Based on our case, we suggest that BAL with immunohistochemical staining can be a valuable modality to eliminate the possibility of infection and other infiltrating disorders, and diagnose pulmonary LCH in case of suspicious pulmonary lesions.
Keywords: Pulmonary, Langerhans cell histiocytosis, Bronchoalveolar lavage, CD1a
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October 2017, 24 (2)
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