Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 6, 2022; 10(4): 1423-1431
Published online Feb 6, 2022. doi: 10.12998/wjcc.v10.i4.1423
Langerhans cell histiocytosis presenting as an isolated brain tumour: A case report
Han-Xiang Liang, Yue-Long Yang, Qing Zhang, Zhi Xie, En-Tao Liu, Shu-Xia Wang
Han-Xiang Liang, Department of Nuclear Medicine, Maoming People's Hospital, Maoming 525000, Guangdong Province, China
Yue-Long Yang, Department of Radiology, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangzhou 510080, Guangdong Province, China
Qing Zhang, En-Tao Liu, Shu-Xia Wang, WeiLun PET Center, Department of Nuclear Medicine, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangzhou 510080, Guangdong Province, China
Zhi Xie, Guangdong Lung Cancer Institute, Guangdong Provincial Key Laboratory of Translational Medicine in Lung Cancer, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Guangzhou 510080, Guangdong Province, China
Author contributions: Liang HX and Yang YL contributed to the conception and design; Liang HX, Yang YL, and Zhang Q contributed to the interpretation of imaging; Yang YL, Zhang Q, and Xie Z contributed to the interpretation of history, treatments, and histopathology; Liang HX and Liu ET contributed to the drafting of the manuscript; Liu ET and Wang SX contributed to the revised the manuscript.
Supported by Guangdong Medical Research Fund to Han-Xiang Liang, No. B2021084; Traditional Chinese Medicine Bureau of Guangdong Province to En-Tao Liu, No. 20211005; and High-level Hospital Construction Research Project of Maoming People's Hospital to Han-Xiang Liang, No. ZX2020014.
Informed consent statement: Publication consent has been obtained from the patient in a written form.
Conflict-of-interest statement: All authors have completed the ICMJE uniform disclosure form. The authors have no conflicts of interest 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 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: En-Tao Liu, MD, Doctor, WeiLun PET Center, Department of Nuclear Medicine, Guangdong Provincial People's Hospital, Guangdong Academy of Medical Sciences, Room 526, 5/F, Weilun Building of Guangdong Provincial People's Hospital, No. 106 Zhongshan Er Road, Guangzhou 510080, Guangdong Province, China. liuentao@gdph.org.cn
Received: September 5, 2021
Peer-review started: September 5, 2021
First decision: October 22, 2021
Revised: November 3, 2021
Accepted: December 23, 2021
Article in press: December 23, 2021
Published online: February 6, 2022
Processing time: 141 Days and 5.8 Hours
Abstract
BACKGROUND

Langerhans cell histiocytosis (LCH) is a rare proliferative histiocyte disorder. It can affect any organ or system, especially the bone, skin, lung, and central nervous system (CNS). In the CNS, the hypothalamic-pituitary is predominantly affected, whereas the brain parenchyma is rarely affected. LCH occurring in the brain parenchyma can be easily confused with glioblastoma or brain metastases. Thus, multimodal imaging is useful for the differential diagnosis of these intracerebral lesions and detection of lesions in the other organs.

CASE SUMMARY

A 47-year-old man presented with a headache for one week and sudden syncope. Brain computed tomography (CT) and magnetic resonance imaging showed an irregularly shaped nodule with heterogeneous enhancement. On 18F-fluorodeoxyglucose (18F-FDG) positron emission tomography/CT, a nodule with 18F-FDG uptake and multiple cysts in the upper lobes of both lungs were noted, which was also confirmed by high-resolution CT. Thus, the patient underwent surgical resection of the brain lesion for further examination. Postoperative pathology confirmed LCH. The patient received chemotherapy after surgery. No recurrence was observed in the brain at the 12-mo follow-up.

CONCLUSION

Multimodal imaging is useful for evaluating the systemic condition of LCH, developing treatment plans, and designing post-treatment strategies.

Keywords: Langerhans cell histiocytosis; Brain neoplasms; Lung; Computed tomography; Magnetic resonance imaging; Positron emission tomography/computed tomography; Case report

Core Tip: Langerhans cell histiocytosis (LCH) is a rare hematological disease characterized by a clonal proliferation of abnormal langerhans cells. It can affect any organ or system, especially the bone, skin, lung, and central nervous system (CNS). In the CNS, the hypothalamic-pituitary is predominantly affected, whereas the brain parenchyma is rarely affected. Cases of LCH involving the brain parenchyma and presenting as an isolated brain tumour have been reported, but all the reports lack complete multimodal imaging. In this manuscript, we have reported a case of LCH involving the brain parenchyma and bilateral lungs, which was assessed using computed tomography (CT), high-resolution CT, magnetic resonance imaging, and 18F-fluorodeoxyglucose positron emission tomography/CT. Furthermore, we have reviewed the relevant literature.