ArticleOxford medical case reports2025
Multisystem Langerhans cell Histiocytosis presenting with spontaneous pneumothorax in a toddler: case report and literature review.
Article in Oxford medical case reports, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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Abstract
Multisystem Langerhans cell histiocytosis (MS-LCH) is exceedingly rare in very young children and can present with predominant lung involvement. We report a 2-year-old boy who presented with prolonged fever, cough, and acute respiratory distress. Chest imaging revealed recurrent bilateral pneumothoraces requiring multiple chest drains; high-resolution computed tomography showed diffuse bilateral thin-walled lung cysts. Thoracoscopic lung biopsy with immunohistochemistry (CD1a+, CD45+, S100+) confirmed Langerhans cell histiocytosis. Given the strong association between pediatric pulmonary LCH and multisystem disease, a whole-body FDG-PET/CT was performed, revealing hepatic and splenic involvement and confirming a diagnosis of MS-LCH. Systemic chemotherapy with vinblastine and corticosteroids led to clinical improvement. We also review nine similar pediatric cases reported in the literature, highlighting the importance of recognizing pulmonary findings as a gateway to diagnosing underlying multisystem LCH.
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