Chen C, Huang XL, Gao DQ, Li YW, Qian SX. Chronic myelomonocytic leukemia-associated pulmonary alveolar proteinosis: A case report and review of literature. World J Clin Cases 2021; 9(5): 1156-1167 [PMID: 33644180 DOI: 10.12998/wjcc.v9.i5.1156]
Corresponding Author of This Article
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
Research Domain of This Article
Hematology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
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/
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 byThe 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 Processing time: 106 Days and 8.1 Hours
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.
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.
