Pseudomonas aeruginosa sepsis presenting as oral ecthyma gangrenosum in identical twins with Bruton tyrosine kinase gene mutation: Two case reports and review of the literature

Sheng-Chieh Lin; Bor-Luen Chiang; Yuarn-Jang Lee; Ya-Ting Chang; Shiuh-Bin Fang

doi:10.1016/j.jmii.2020.04.009

Pseudomonas aeruginosa sepsis presenting as oral ecthyma gangrenosum in identical twins with Bruton tyrosine kinase gene mutation: Two case reports and review of the literature

Sheng-Chieh Lin
, Bor-Luen Chiang
, Yuarn-Jang Lee
, Ya-Ting Chang
, Shiuh-Bin Fang

Research output: Contribution to journal › Article › peer-review

7 Citations (Scopus)

Abstract

X-linked agammaglobulinemia (XLA) is a primary immunodeficiency disease. We reported two 7-month-old identical male twins with Pseudomonas aeruginosa sepsis who initially manifested as oral ecthyma gangrenosum and were finally diagnosed to have XLA. In both cases, we confirmed the c.862C > T BTK missense mutation in exon 10 at the SH2 domain.

Original language	English
Pages (from-to)	1030-1034
Number of pages	5
Journal	Chinese Journal of Microbiology and Immunology
Volume	53
Issue number	6
Early online date	Apr 17 2020
DOIs	https://doi.org/10.1016/j.jmii.2020.04.009
Publication status	Published - Dec 2020

Keywords

Bruton tyrosine kinase
Ecthyma gangrenosum
Identical twins
Pseudomonas aeruginosa sepsis
X-linked agammaglo bulinemia

ASJC Scopus subject areas

Microbiology (medical)
Infectious Diseases
General Immunology and Microbiology
Immunology and Allergy

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10.1016/j.jmii.2020.04.009

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Pseudomonas aeruginosa sepsis presenting as oral ecthyma gangrenosum in identical twins with Bruton tyrosine kinase gene mutation: Two case reports and review of the literature. / Lin, Sheng-Chieh; Chiang, Bor-Luen; Lee, Yuarn-Jang et al.
In: Chinese Journal of Microbiology and Immunology, Vol. 53, No. 6, 12.2020, p. 1030-1034.

Research output: Contribution to journal › Article › peer-review

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abstract = "X-linked agammaglobulinemia (XLA) is a primary immunodeficiency disease. We reported two 7-month-old identical male twins with Pseudomonas aeruginosa sepsis who initially manifested as oral ecthyma gangrenosum and were finally diagnosed to have XLA. In both cases, we confirmed the c.862C > T BTK missense mutation in exon 10 at the SH2 domain.",

keywords = "Bruton tyrosine kinase, Ecthyma gangrenosum, Identical twins, Pseudomonas aeruginosa sepsis, X-linked agammaglo bulinemia",

author = "Sheng-Chieh Lin and Bor-Luen Chiang and Yuarn-Jang Lee and Ya-Ting Chang and Shiuh-Bin Fang",

note = "Funding Information: We thank Dr. Yin-Hsiu Chien and Hsin-Hui Yu for their assistance in the mutation study of the BTK gene, Dr. Suei-Tsau Tsai for his referral of the patients, and Dr. Ke-Chung Chang for his surgical care. Publisher Copyright: {\textcopyright} 2020",

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AU - Lee, Yuarn-Jang

AU - Chang, Ya-Ting

AU - Fang, Shiuh-Bin

N1 - Funding Information: We thank Dr. Yin-Hsiu Chien and Hsin-Hui Yu for their assistance in the mutation study of the BTK gene, Dr. Suei-Tsau Tsai for his referral of the patients, and Dr. Ke-Chung Chang for his surgical care. Publisher Copyright: © 2020

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AB - X-linked agammaglobulinemia (XLA) is a primary immunodeficiency disease. We reported two 7-month-old identical male twins with Pseudomonas aeruginosa sepsis who initially manifested as oral ecthyma gangrenosum and were finally diagnosed to have XLA. In both cases, we confirmed the c.862C > T BTK missense mutation in exon 10 at the SH2 domain.

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KW - Ecthyma gangrenosum

KW - Identical twins

KW - Pseudomonas aeruginosa sepsis

KW - X-linked agammaglo bulinemia

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IS - 6

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Pseudomonas aeruginosa sepsis presenting as oral ecthyma gangrenosum in identical twins with Bruton tyrosine kinase gene mutation: Two case reports and review of the literature

Abstract

Keywords

ASJC Scopus subject areas

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