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Yamazaki S, Ohtsuka Y, Yokokura T, et al. Eosinophilic gastroenteritis in a patient with Bruton's tyrosine kinase deficiency. Pediatr Int. 2016;58(5):417-419doi: 10.1111/ped.12823.
Yamazaki, S., Ohtsuka, Y., Yokokura, T., Yokota, R., Honjo, A., Inage, E., Baba, Y., Mori, M., Suzuki, R., Iwata, T., & Shimizu, T. (2016). Eosinophilic gastroenteritis in a patient with Bruton's tyrosine kinase deficiency. Pediatrics international : official journal of the Japan Pediatric Society, 58(5), 417-419. https://doi.org/10.1111/ped.12823
Yamazaki, Susumu, et al. "Eosinophilic gastroenteritis in a patient with Bruton's tyrosine kinase deficiency." Pediatrics international : official journal of the Japan Pediatric Society vol. 58,5 (2016): 417-419. doi: https://doi.org/10.1111/ped.12823
Yamazaki S, Ohtsuka Y, Yokokura T, Yokota R, Honjo A, Inage E, Baba Y, Mori M, Suzuki R, Iwata T, Shimizu T. Eosinophilic gastroenteritis in a patient with Bruton's tyrosine kinase deficiency. Pediatr Int. 2016 May;58(5):417-419. doi: 10.1111/ped.12823. Epub 2016 Feb 03. PMID: 26840762.
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Pediatr Int. 2016 May;58(5):417-419. doi: 10.1111/ped.12823. Epub 2016 Feb 03.

Eosinophilic gastroenteritis in a patient with Bruton's tyrosine kinase deficiency.

Pediatrics international : official journal of the Japan Pediatric Society

Susumu Yamazaki, Yoshikazu Ohtsuka, Tomoaki Yokokura, Rena Yokota, Asuka Honjo, Eisuke Inage, Yosuke Baba, Mari Mori, Ryuyo Suzuki, Tsutomu Iwata, Toshiaki Shimizu

Affiliations

  1. Department of Pediatrics and Adolescent Medicine, Juntendo University Graduate School of Medicine, Tokyo, Japan.
  2. Pediatric Science, Department of Child Health and Development, Faculty of Home Economics, Tokyo Kasei University, Tokyo, Japan.

PMID: 26840762 DOI: 10.1111/ped.12823

Abstract

Eosinophilic gastrointestinal diseases (EGID) are relatively rare diseases characterized by eosinophilic infiltration of the gastrointestinal tract resulting in various gastrointestinal symptoms. EGID are often caused by allergic reactions or systemic eosinophilic disorders, but their comorbidity with Bruton's tyrosine kinase (BTK) deficiency has not been previously documented. Here, we report a case of eosinophilic gastroenteritis (EG) in a patient with BTK deficiency. Despite adequate replacement immunoglobulin (Ig) therapy, trough serum IgG was not maintained. To identify the underlying cause of the low trough level and chronic diarrhea, the intestine was investigated on endoscopy. We also screened for the variable number of tandem repeat polymorphism in FCGRT. Genetic analysis could not explain the low trough IgG, but endoscopy indicated eosinophilic enterocolitis. EG may be an important differential diagnosis when primary immunodeficiency patients have chronic diarrhea or continued low serum IgG.

© 2016 Japan Pediatric Society.

Keywords: Bruton's agammaglobulinemia; FCGRT; X-linked agammaglobulinemia; gastroenteritis; neonatal Fc receptor (FcRn)

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