Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 16, 2021; 9(5): 1156-1167
Published online Feb 16, 2021. doi: 10.12998/wjcc.v9.i5.1156
Chronic myelomonocytic leukemia-associated pulmonary alveolar proteinosis: A case report and review of literature
Can Chen, Xi-Lian Huang, Da-Quan Gao, Yi-Wei Li, Shen-Xian Qian
Can Chen, Xi-Lian Huang, Da-Quan Gao, Shen-Xian Qian, Department of Hematology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
Yi-Wei Li, Department of Intensive Care Unit, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, Hangzhou 310006, Zhejiang Province, China
Author contributions: Huang XL proposed the research idea; Chen C, Gao DQ and Qian SX were involved in the case management; Chen C, Gao DQ and Li YW performed the literature search and selection; Huang XL, Chen C and Li YW drafted the manuscript; Huang XL and Qian SX critically reviewed the manuscript; All authors approved the final version of the manuscript.
Supported by The Medical and Health Technology Project of Zhejiang Province, No. 2018KY567; and the Hangzhou Municipal Science and Technology Development Program, No. 20142013A61.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have 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: Xi-Lian Huang, MD, PhD, Doctor, Department of Hematology, Affiliated Hangzhou First People’s Hospital, Zhejiang University School of Medicine, No. 216 Huansha Road, Hangzhou 310006, Zhejiang Province, China. syxlhuang@163.com
Received: October 16, 2020
Peer-review started: October 16, 2020
First decision: November 24, 2020
Revised: December 6, 2020
Accepted: December 16, 2020
Article in press: December 16, 2020
Published online: February 16, 2021
Abstract
BACKGROUND

Pulmonary alveolar proteinosis (PAP) is a rare condition that can cause progressive symptoms including dyspnea, cough and respiratory insufficiency. Secondary PAP is generally associated with hematological malignancies including chronic myelomonocytic leukemia (CMML). To the best of our knowledge, this is the first reported case of PAP occurring secondary to CMML.

CASE SUMMARY

We report the case of a 63-year-old male who presented with a recurrent cough and gradually progressive dyspnea in the absence of fever. Based upon clinical symptoms, computed tomography findings, bone marrow aspiration, flow cytometry studies and cytogenetic analyses, the patient was diagnosed with PAP secondary to CMML. He underwent whole lung lavage in March 2016 to alleviate his dyspnea, after which he began combined chemotherapeutic treatment with decitabine and cytarabine. The patient died in January 2020 as a consequence of severe pulmonary infection.

CONCLUSION

This case offers insight regarding the mechanistic basis for PAP secondary to CMML and highlights potential risk factors.

Keywords: Pulmonary alveolar proteinosis, Chronic myelomonocytic leukemia, Diagnosis, Treatment, Prognosis, Case report

Core Tip: Pulmonary alveolar proteinosis secondary to chronic myelomonocytic leukemia is a rare disease that has been insufficiently studied to date. Herein, we report a case of pulmonary alveolar proteinosis secondary to chronic myelomonocytic leukemia and perform a comprehensive literature review. We also provide a framework for understanding the mechanistic basis of this disease. It is likely that pulmonary alveolar proteinosis symptoms can be alleviated by achieving primary disease control. Together, these findings may contribute to improvements in clinical practice.