論文

国際誌
2021年

Case Report: Analysis of Preserved Umbilical Cord Clarified X-Linked Anhidrotic Ectodermal Dysplasia With Immunodeficiency in Deceased, Undiagnosed Uncles.

Frontiers in immunology
  • Satoshi Inaba
  • ,
  • Yuta Aizawa
  • ,
  • Yuki Miwa
  • ,
  • Chihaya Imai
  • ,
  • Hidenori Ohnishi
  • ,
  • Hirokazu Kanegane
  • ,
  • Akihiko Saitoh

12
開始ページ
786164
終了ページ
786164
記述言語
英語
掲載種別
研究論文(学術雑誌)
DOI
10.3389/fimmu.2021.786164

Family history is one key in diagnosing inborn errors of immunity (IEI); however, disease status is difficult to determine in deceased relatives. X-linked anhidrotic ectodermal dysplasia with immunodeficiency is one of the hyper IgM syndromes that is caused by a hypomorphic variant in the nuclear factor kappa beta essential modulator. We identified a novel IKBKG variant in a 7-month-old boy with pneumococcal rib osteomyelitis and later found that his mother has incontinentia pigmenti. Genetic analysis of preserved umbilical cords revealed the same variant in two of his deceased maternal uncles. Analysis of preserved umbilical cord tissue from deceased relatives can provide important information for diagnosing IEI in their descendants.

リンク情報
DOI
https://doi.org/10.3389/fimmu.2021.786164
PubMed
https://www.ncbi.nlm.nih.gov/pubmed/35003103
PubMed Central
https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8727435
ID情報
  • DOI : 10.3389/fimmu.2021.786164
  • PubMed ID : 35003103
  • PubMed Central 記事ID : PMC8727435

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