Zhang T, Li M, Tan L, Li X. Pulmonary alveolar proteinosis induced by X-linked agammaglobulinemia: A case report. World J Clin Cases 2024; 12(9): 1644-1648 [PMID: 38576739 DOI: 10.12998/wjcc.v12.i9.1644]
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. Mar 26, 2024; 12(9): 1644-1648 Published online Mar 26, 2024. doi: 10.12998/wjcc.v12.i9.1644
Pulmonary alveolar proteinosis induced by X-linked agammaglobulinemia: A case report
Ting Zhang, Ming Li, Li Tan, Xin Li
Ting Zhang, Ming Li, Li Tan, Department of Respiratory Medicine, Kunming Children´s Hospital, Kunming 650228, Yunnan Province, China
Xin Li, Department of Emergency, Kunming Children's Hospital, Kunming 650228, Yunnan Province, China
Co-first authors: Ting Zhang and Ming Li.
Author contributions: Zhang T managed the case, wrote and corrected the manuscript; Li X assisted with writing, correction, and reconstruction of the manuscript; All authors have read and approved the final manuscript.
Supported bySanitation Research Project of Kunming Municipal Health Commission, No. 2020-06-01-119.
Informed consent statement: Written informed consent was obtained from the patient. A copy of the written consent is available for review from the Editor of this journal.
Conflict-of-interest statement: The authors declare that they have no competing interests.
CARE Checklist (2016) statement: The authors have read the CARE Checklist statement, and the manuscript was prepared and revised according to the CARE Checklist statement.
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: https://creativecommons.org/Licenses/by-nc/4.0/
Received: October 20, 2023 Peer-review started: October 20, 2023 First decision: January 5, 2024 Revised: January 20, 2024 Accepted: March 1, 2024 Article in press: March 1, 2024 Published online: March 26, 2024 Processing time: 156 Days and 23.7 Hours
Abstract
BACKGROUND
Pulmonary alveolar proteinosis (PAP) and X-linked agammaglobulinemia (XLA) are rare diseases in children. Many theories infer that immunodeficiency can induce PAP, but these reports are almost all review articles, and there is little clinical evidence. We report the case of a child with both PAP and XLA.
CASE SUMMARY
A 4-month-old boy sought medical treatment due to coughing and difficulty in breathing for > 2 wk. He had been hospitalized multiple times due to respiratory infections and diarrhea. Chest computed tomography and alveolar lavage fluid showed typical PAP-related manifestations. Genetic testing confirmed that the boy also had XLA. Following total lung alveolar lavage and intravenous immunoglobulin replacement therapy, the boy recovered and was discharged. During the follow-up period, the number of respiratory infections was significantly reduced, and PAP did not recur.
CONCLUSION
XLA can induce PAP and improving immune function contributes to the prognosis of children with this type of PAP.
Core Tip: Pulmonary alveolar proteinosis (PAP) and X-linked agammaglobulinemia (XLA) are both rare diseases in children. This article shares the diagnosis and treatment process of a special case to confirm that XLA was a secondary cause of PAP which improved with intravenous immunoglobulin treatment.
