Extracorporeal Membrane Oxygenation-Dependent Fulminant Melioidosis From Caspase 4 Mutation Reversed by Interferon Gamma Therapy

被引:5
作者
Amali, Aseervatham Anusha [1 ]
Ravikumar, Sharada [1 ]
Chew, Wei Leong [2 ,3 ]
Tan, Zhaohong [1 ]
Sam, Qi Hui [1 ,3 ]
Chen, Kaiwen W. [4 ,5 ]
Boucher, Dave [6 ]
MacLaren, Graeme [7 ,8 ]
Chai, Louis Yi Ann [1 ,3 ,9 ]
机构
[1] Natl Univ Hlth Syst, Dept Med, Div Infect Dis, NUHS Tower Block,1E Kent Ridge Rd, Singapore 119228, Singapore
[2] Agcy Sci Technol & Res, Genome Inst Singapore, Singapore, Singapore
[3] Natl Univ Singapore, Synthet Biol Clin & Technol Innovat, Singapore, Singapore
[4] Natl Univ Singapore, Life Sci Inst, Immunol Programme, Singapore, Singapore
[5] Natl Univ Singapore, Yong Loo Lin Sch Med, Dept Microbiol & Immunol, Immunol Translat Res Programme, Singapore, Singapore
[6] Univ York, York Biomed Res Inst, Dept Biol, York, England
[7] Natl Univ Singapore, Yong Loo Lin Sch Med, Dept Surg, Singapore, Singapore
[8] Natl Univ Singapore Hosp, Natl Univ Heart Ctr, Cardiothorac Intens Care Unit, Singapore, Singapore
[9] Natl Univ Singapore, Yong Loo Lin Sch Med, Dept Med, Singapore, Singapore
关键词
Burkholderia pseudomallei; extracorporeal membrane oxygenation; exome sequencing; inflammasome; immunotherapy; INFLAMMATORY CASPASES; GASDERMIN D; ACTIVATION; GSDMD;
D O I
10.1093/cid/ciad517
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
We describe bedside-to-bench immunological and genetic elucidation of defective pyroptosis attributable to novel caspase 4 defect mediating pathogen-triggered inflammatory programmed cell death, in the setting of severe pneumonia and abscess-forming melioidosis in an overtly healthy host failing to clear Burkholderia pseudomallei infection, and how targeted adjunctive biological therapy led to a successful outcome.
引用
收藏
页码:94 / 97
页数:4
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