Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 26, 2019; 7(14): 1892-1898
Published online Jul 26, 2019. doi: 10.12998/wjcc.v7.i14.1892
Pulmonary Langerhans cell histiocytosis in adults: A case report
Feng-Feng Wang, Ya-Shuang Liu, Wei-Bo Zhu, Yan-Dong Liu, Yao Chen
Feng-Feng Wang, Ya-Shuang Liu, Wei-Bo Zhu, Yan-Dong Liu, Yao Chen, Department of Respiration, People’s Hospital of Jingjiang, YangZhou University Medical Academy, Jingjiang 214500, Jiangsu Province, China
Author contributions: Wang FF designed the report; Zhu WB and Liu YD collected the patient’s clinical data and performed the tracheoscopy procedure; Liu YS analysed the imaging report; Chen Y revised the manuscript for intellectual content; Wang FF was a major contributor in writing of the manuscript; all the authors approved the final version of the article to be published.
Informed consent statement: Consent was obtained from the patient for publication of this report and accompanying images.
Conflict-of-interest statement: The authors have no conflict of interests to declare.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Yao Chen, MD, Doctor, Department of Respiration, People’s Hospital of Jingjiang, YangZhou University Medical Academy, No. 28 Zhongzhou Road, Jingjiang 214500, Jiangsu Province, China. chenyao760@outlook.com
Telephone: +86-523-84996381Fax: +86-523-84996381
Received: April 8, 2019
Peer-review started: April 8, 2019
First decision: June 12, 2019
Revised: June 16, 2019
Accepted: June 20, 2019
Article in press: June 21, 2019
Published online: July 26, 2019
Processing time: 110 Days and 11.4 Hours
Abstract
BACKGROUND

Langerhans cell histiocytosis (LCH) is a rare disease of unknown aetiology. While it may affect any organ of the body, few cases of solitary lung involvement are published in the literature. Here, we report a rare case of pulmonary LCH (PLCH) in an adult.

CASE SUMMARY

A 52-year-old male presented to hospital in July 2018 with complaints of progressively worsening cough with sputum, breathlessness, easy fatigability, and loss of appetite since 2016, and a 32-year history of heavy cigarette smoking (average 30 cigarettes/d). Physical examination showed only weakened breathing sounds and wheezing during lung auscultation. Chest computed tomography (CT) showed irregular micronodules and multiple thin-walled small holes. Respiratory function tests showed a slight decrease. Ultrasonic cardiogram showed mild tricuspid regurgitation and no pulmonary hypertension. Fibreoptic bronchoscopy was performed with transbronchial biopsies from the basal segment of right lower lobe. LCH was confirmed by immunohistochemistry. The final diagnosis was PLCH without extra-pulmonary involvement. We suggested smoking cessation treatment. A 3-mo follow-up chest CT scan showed clear absorption of the nodule and thin-walled small holes. The symptoms of cough and phlegm had improved markedly and appetite had improved. There was no obvious dyspnoea.

CONCLUSION

Imaging manifestations of nodules, cavitating nodules, and thick-walled or thin-walled cysts prompted suspicion of PLCH and lung biopsy for diagnosis.

Keywords: Pulmonary Langerhans’ cell histiocytosis; Adult; Smoking cessation; Imaging; Nodules; Case report

Core tip: Langerhans cell histiocytosis (LCH) is a rare disorder of unknown aetiology and may affect any organ of the body. Local LCH only affects the lungs and is known as pulmonary LCH (PLCH). Reports of PLCH cases are relatively rare in the literature. This report describes the presentation, clinical investigation and diagnosis of a PLCH case involving a 52-year-old male with a 1-year history of progressively worsening cough with sputum, breathlessness, easy fatigability, and loss of appetite, and 32-yr history of heavy cigarette smoking. We also discuss the clinical presentation and signs, diagnosis, treatment, and outcome of such rare cases.