From diagnostic biopsy to bullectomy: A case of progressive complications in pulmonary langerhans cell histiocytosis

A 38-year-old male smoker presented with a persistent dry cough lasting several weeks. Chest radiography showed bilateral reticulonodular opacities predominantly in the upper lung fields. Subsequent chest computed tomography revealed diffuse, thick-walled, irregular cystic lesions mainly involving the mid and upper lung zones with a centrilobular distribution. Histopathological examination obtained through video-assisted thoracoscopic surgery demonstrated bronchiolocentric nodules and cysts containing Langerhans cells with convoluted nuclei, confirmed by positive immunohistochemical staining for CD1a, S-100, and Langerin. These findings confirmed the diagnosis of pulmonary Langerhans Cell Histiocytosis (PLCH). The patient's symptoms improved initially following smoking cessation but were complicated by recurrent pneumothorax and the formation of giant bullae, ultimately requiring surgical intervention. This case emphasizes the association between PLCH and smoking, highlights potential complications following surgical biopsy, and underscores smoking cessation as a critical therapeutic measure, with targeted therapies reserved for refractory or mutation-specific cases.

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Publication Details

Journal
Respiratory Medicine Case Reports
Published
2025-01-01
DOI
https://doi.org/10.1016/j.rmcr.2025.102263
Primary Topic
Histiocytic Disorders and Treatments
Type
article
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article

From diagnostic biopsy to bullectomy: A case of progressive complications in pulmonary langerhans cell histiocytosis

Yung‐Chia Huang, Pang‐Kuo Lo, Yen‐Lin Wu, Chih‐yu Shih et al.
Respiratory Medicine Case Reports
Histiocytic Disorders and Treatments
article

From diagnostic biopsy to bullectomy: A case of progressive complications in pulmonary langerhans cell histiocytosis

Yung‐Chia Huang, Pang‐Kuo Lo, Yen‐Lin Wu, Chih‐yu Shih, Yuan-Chia Hsin
article en

Abstract

A 38-year-old male smoker presented with a persistent dry cough lasting several weeks. Chest radiography showed bilateral reticulonodular opacities predominantly in the upper lung fields. Subsequent chest computed tomography revealed diffuse, thick-walled, irregular cystic lesions mainly involving the mid and upper lung zones with a centrilobular distribution. Histopathological examination obtained through video-assisted thoracoscopic surgery demonstrated bronchiolocentric nodules and cysts containing Langerhans cells with convoluted nuclei, confirmed by positive immunohistochemical staining for CD1a, S-100, and Langerin. These findings confirmed the diagnosis of pulmonary Langerhans Cell Histiocytosis (PLCH). The patient's symptoms improved initially following smoking cessation but were complicated by recurrent pneumothorax and the formation of giant bullae, ultimately requiring surgical intervention. This case emphasizes the association between PLCH and smoking, highlights potential complications following surgical biopsy, and underscores smoking cessation as a critical therapeutic measure, with targeted therapies reserved for refractory or mutation-specific cases.

Respiratory Medicine Case ReportsVol. 57
National Yang Ming Chiao Tung University (TW), Ministry of Health and Welfare (TW)
Good health and well-being
Openalex Percentile: Top 76%
Histiocytic Disorders and Treatments
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From diagnostic biopsy to bullectomy: A case of progressive complications in pulmonary langerhans cell histiocytosis — Yung‐Chia Huang, Pang‐Kuo Lo, et al. · Respiratory Medicine Case Reports (2025) | TGRS Research Map | TGRS