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Pulmonary Alveolar Proteinosis Due to Pneumocystis carinii in Type 1 Hyper-IgM Syndrome: A Case Report.
Front Pediatr. 2020; 8:264.FP

Abstract

Background:

Pulmonary alveolar proteinosis (PAP) is a rare diffuse lung disease. Reports of rare cases of PAP due to Pneumocystis jirovecii (P. jirovecii) exist in infants with immunodeficiency diseases, but no cases have been reported to date in pediatric patients with type 1 hyper-IgM syndrome (HIGM1). Case Presentation: Herein, we present a case of PAP secondary to P. jirovecii on an infant with HIGM1. He was admitted to our unit because of cough and tachypnea. Lung biopsy confirmed the diagnosis of PAP, whereas hexamine-silver staining of the bronchoalveolar lavage fluid identified P. jirovecii infection. No other probable cause of PAP was observed. Whole exome sequencing indicated a novel c.511dupA (p.I171N*30) hemizygous mutation in the CD40 ligand (CD40LG) gene. He was cured with bronchoalveolar lavage and compound sulfamethoxazole tablets.

Conclusions:

To our knowledge, this is the first reported case of P. jirovecii infection as a reversible cause of PAP in an infant with HIGM1.

Authors+Show Affiliations

Department of Pneumology, Zhejiang University School of Medicine of Children's Hospital, Hangzhou, China.Department of Pneumology, Zhejiang University School of Medicine of Children's Hospital, Hangzhou, China.Department of Pneumology, Zhejiang University School of Medicine of Children's Hospital, Hangzhou, China.Department of Pneumology, Zhejiang University School of Medicine of Children's Hospital, Hangzhou, China.

Pub Type(s)

Case Reports

Language

eng

PubMed ID

32596190

Citation

Zhang, Fei Zhou, et al. "Pulmonary Alveolar Proteinosis Due to Pneumocystis Carinii in Type 1 Hyper-IgM Syndrome: a Case Report." Frontiers in Pediatrics, vol. 8, 2020, p. 264.
Zhang FZ, Yuan JX, Qin L, et al. Pulmonary Alveolar Proteinosis Due to Pneumocystis carinii in Type 1 Hyper-IgM Syndrome: A Case Report. Front Pediatr. 2020;8:264.
Zhang, F. Z., Yuan, J. X., Qin, L., & Tang, L. F. (2020). Pulmonary Alveolar Proteinosis Due to Pneumocystis carinii in Type 1 Hyper-IgM Syndrome: A Case Report. Frontiers in Pediatrics, 8, 264. https://doi.org/10.3389/fped.2020.00264
Zhang FZ, et al. Pulmonary Alveolar Proteinosis Due to Pneumocystis Carinii in Type 1 Hyper-IgM Syndrome: a Case Report. Front Pediatr. 2020;8:264. PubMed PMID: 32596190.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Pulmonary Alveolar Proteinosis Due to Pneumocystis carinii in Type 1 Hyper-IgM Syndrome: A Case Report. AU - Zhang,Fei Zhou, AU - Yuan,Jie Xin, AU - Qin,Lu, AU - Tang,Lan Fang, Y1 - 2020/06/11/ PY - 2020/01/17/received PY - 2020/04/27/accepted PY - 2020/6/30/entrez PY - 2020/7/1/pubmed PY - 2020/7/1/medline KW - CD40 ligand (CD40LG) KW - Pneumocystis jirovecii (P. jirovecii) KW - hyper IgM syndrome KW - infant KW - pulmonary alveolar proteinosis (PAP) KW - type 1 (HIGM1) SP - 264 EP - 264 JF - Frontiers in pediatrics JO - Front Pediatr VL - 8 N2 - Background: Pulmonary alveolar proteinosis (PAP) is a rare diffuse lung disease. Reports of rare cases of PAP due to Pneumocystis jirovecii (P. jirovecii) exist in infants with immunodeficiency diseases, but no cases have been reported to date in pediatric patients with type 1 hyper-IgM syndrome (HIGM1). Case Presentation: Herein, we present a case of PAP secondary to P. jirovecii on an infant with HIGM1. He was admitted to our unit because of cough and tachypnea. Lung biopsy confirmed the diagnosis of PAP, whereas hexamine-silver staining of the bronchoalveolar lavage fluid identified P. jirovecii infection. No other probable cause of PAP was observed. Whole exome sequencing indicated a novel c.511dupA (p.I171N*30) hemizygous mutation in the CD40 ligand (CD40LG) gene. He was cured with bronchoalveolar lavage and compound sulfamethoxazole tablets. Conclusions: To our knowledge, this is the first reported case of P. jirovecii infection as a reversible cause of PAP in an infant with HIGM1. SN - 2296-2360 UR - https://www.unboundmedicine.com/medline/citation/32596190/Pulmonary_Alveolar_Proteinosis_Due_to_Pneumocystis_carinii_in_Type_1_Hyper-IgM_Syndrome:_A_Case_Report L2 - https://www.ncbi.nlm.nih.gov/pmc/articles/pmid/32596190/ DB - PRIME DP - Unbound Medicine ER -
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