Case Report
Copyright ©The Author(s) 2019. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 6, 2019; 7(17): 2519-2525
Published online Sep 6, 2019. doi: 10.12998/wjcc.v7.i17.2519
Characteristics of multiple nodules in a patient with pulmonary Langerhans cell histiocytosis: A case report
Nobuhiro Kanaji, Yoshimasa Tokunaga, Ryou Ishikawa, Naoki Watanabe, Norimitsu Kadowaki
Nobuhiro Kanaji, Naoki Watanabe, Norimitsu Kadowaki, Department of Internal Medicine, Division of Hematology, Rheumatology and Respiratory Medicine, Faculty of Medicine, Kagawa University, Kagawa 761-0793, Japan
Yoshimasa Tokunaga, Department of General Thoracic Surgery, Breast and Endocrinological Surgery, Faculty of Medicine, Kagawa University, Kagawa 761-0793, Japan
Ryou Ishikawa, Department of Diagnostic Pathology, Faculty of Medicine, Kagawa University, Kagawa 761-0793, Japan
Author contributions: Kanaji N carried out the diagnosis and followed up the patient, designed and drafted the manuscript; Tokunaga Y carried out the diagnosis; Ishikawa R collected pathological sections; Watanabe N and Kadowaki N revised the manuscript; all authors read and approved the final manuscript.
Informed consent statement: Written informed consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: No potential conflicts of interest relevant to this article were reported.
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: Nobuhiro Kanaji, MD, PhD, Assistant Professor, Department of Internal Medicine, Division of Hematology, Rheumatology and Respiratory Medicine, Faculty of Medicine, Kagawa University, 1750-1 Ikenobe, Kagawa 761-0793, Japan. kanaji@med.kagawa-u.ac.jp
Telephone: +81-87-8912145 Fax: +81-87-8912147
Received: April 17, 2019
Peer-review started: April 18, 2019
First decision: June 19, 2019
Revised: July 5, 2019
Accepted: July 20, 2019
Article in press: July 20, 2019
Published online: September 6, 2019
Processing time: 143 Days and 10.2 Hours
Abstract
BACKGROUND

The common computed tomography findings of pulmonary Langerhans cell histiocytosis (PLCH) are multiple cysts and micronodules predominantly in middle to upper lung lobes. Non-cystic nodules and large nodules are atypical findings of PLCH.

CASE SUMMARY

The patient was a 48-year-old Japanese man with a smoking history (20 cigarettes/d, 28 years) and no symptoms. Multiple nodules existed in all lung lobes, predominantly in the right lower lobe. Some nodules seemed to be distributed randomly, and others were adjacent to bronchus. Most nodules were solid; some small ones were cystic. The largest nodule was 22 mm in diameter. Although metastatic lung tumors were suspected, thoracoscopic lung biopsy led to the diagnosis of PLCH. At 6 months after he quit smoking, all nodules had almost disappeared. We investigated the characteristics of nodules at diagnosis in detail. Of 349 nodules in total, 116 were in upper and 199 were in lower lobes. Ninety-six (27.5%) were cystic; the remaining 253 (72.5%) were non-cystic. The prevalence of cystic nodules was higher in upper lobes than in lower lobes (right upper 37.5% vs lower 18.2%, P = 0.0068; left upper 48.1% vs lower 24.4%, P = 0.0078). The average size (dia.) of cystic nodules was smaller than that of non-cystic nodules (5.03 mm vs 7.40 mm, respectively, P < 0.0001).

CONCLUSION

Although multiple non-cystic nodules including large nodules (over 20 mm) are atypical, PLCH should be included in differential diagnoses. The presence of small cystic nodules predominantly in upper lobes and asymptomatic situation are also important for differential diagnoses to distinguish from metastatic cancers.

Keywords: Langerhans cell histiocytosis; Multiple; Non-cystic; Nodule; Distribution; Size; Case report

Core tip: The common computed tomography findings of pulmonary Langerhans cell histiocytosis (PLCH) are multiple cysts and micronodules predominantly in middle-to-upper lung lobes. We present herein a rare case of PLCH with non-cystic multiple large nodules. Of 349 nodules in total, 116 were in upper and 199 were in lower lobes. The prevalence of cystic nodules was higher in upper lobes than in lower lobes. The average size (dia.) of cystic nodules was smaller than that of non-cystic nodules. Knowledge of such characteristics of nodules may help the pulmonologists consider PLCH in differential diagnoses of multiple nodules.