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KMID : 0387820170240020157
Clinical Pediatric Hematology-Oncology
2017 Volume.24 No. 2 p.157 ~ p.161
Isolated Pulmonary Langerhans Cell Histiocytosis in an Infant Diagnosed by CD1a Immunostaining of Bronchoalveolar Lavage Cells: A Case Report and Literature Review
Kim Mi-Jin

Yu Jin-Ho
Cho Young-Uk
Yoo Jae-Won
Kang Sung-Han
Kim Hye-Ry
Koh Kyung-Nam
Im Ho-Joon
Seo Jong-Jin
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.
KEYWORD
Pulmonary, Langerhans cell histiocytosis, Bronchoalveolar lavage, CD1a
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