Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Dec 16, 2021; 9(35): 11029-11035
Published online Dec 16, 2021. doi: 10.12998/wjcc.v9.i35.11029
Pulmonary Langerhans cell histiocytosis and multiple system involvement: A case report
Lin Luo, Yan-Xia Li
Lin Luo, Department of Critical Care Medicine, The Second Hospital of Dalian Medical University, Dalian 116023, Liaoning Province, China
Yan-Xia Li, Department of Respiratory and Critical Care Medicine, Institute of Respiratory Diseases, The First Affiliated Hospital of Dalian Medical University, Dalian 116011, Liaoning Province, China
Author contributions: Luo L contributed to data analysis and wrote the paper; Li YX contributed to the conception and design of the study; all authors revised the paper and approved the submitted version.
Informed consent statement: The patient provided informed written consent prior to study enrollment.
Conflict-of-interest statement: There is no conflict-of-interest.
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 that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (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: Yan-Xia Li, PhD, Doctor, Full Professor, Department of Respiratory and Critical Care Medicine, Institute of Respiratory Diseases, The First Affiliated Hospital of Dalian Medical University, No. 222 Zhongshan Road, Dalian 116011, Liaoning Province, China. liyanxia001@163.com
Received: June 1, 2021
Peer-review started: June 1, 2021
First decision: July 15, 2021
Revised: July 15, 2021
Accepted: October 27, 2021
Article in press: October 27, 2021
Published online: December 16, 2021
Abstract
BACKGROUND

Pulmonary Langerhans cell histiocytosis (PLCH) is a relatively rare type of lung disease, common in middle-aged smoking men. It is characterized by proliferation and infiltration of Langerhans cells, and the formation of multiple parabronchial mesenchymal nodules in lung tissue, and may lead to organ dysfunction. There are no typical symptoms and signs, and it is easily misdiagnosed or missed, and therefore deserves clinical attention and further discussion.

CASE SUMMARY

We describe the case of a nonsmoking 46-year-old man with PLCH diagnosed based on clinical manifestations of fever and dry cough, with a history of hypothyroidism and diabetes insipidus for 9 years. Computed tomography (CT)- and CT-guided puncture examinations revealed no abnormalities, and he ultimately underwent thoracoscopic biopsy to confirm the diagnosis. The pathological diagnosis was PLCH. Thyroid function was maintained by medication. Pituitary magnetic resonance imaging showed that the pituitary stalk had become thinner.

CONCLUSION

LCH often involves multiple systems. Moreover, the pathogenesis is not clear, clinical manifestations lack specificity, and diagnosis requires special attention. Diagnosis of PLCH can significantly benefit from comprehensive multidisciplinary analysis.

Keywords: Pulmonary Langerhans cell histiocytosis, Multiple systems, Hypothyroidism, Diabetes insipidus, Case report

Core Tip: We describe the case of a 46-year-old man with pulmonary Langerhans cell histiocytosis, diagnosed due to clinical manifestations of fever and dry cough, with a history of hypothyroidism and diabetes insipidus for 9 years. The pathological diagnosis was lung Langerhans cell histiocytosis. Thyroid function was maintained by medication. Pituitary magnetic resonance imaging showed that the pituitary stalk had become thinner. A comprehensive multidisciplinary analysis can significantly improve disease diagnosis.