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DOI 10.1016/j.bcmd.2021.102596

Медицинская библиотека · Blood cells, molecules & diseases · 2021

Hematologically important mutations: The autosomal forms of chronic granulomatous disease (third update).

Roos Dirk, van Leeuwen Karin, Hsu Amy P, Priel Debra Long, Begtrup Amber, Brandon Rhonda, Rawat Amit, Vignesh Pandiarajan, Madkaikar Manesha, Stasia Marie José, Bakri Faris Ghalib, de Boer Martin, Roesler Joachim, Köker Nezihe, Köker M Yavuz, Jakobsen Marianne, Bustamante Jacinta, Garcia-Morato Maria Bravo, Shephard Juan Luis Valdivieso, Cagdas Deniz, Tezcan Ilhan, Sherkat Roya, Mortaz Esmaeil, Fayezi Abbas, Shahrooei Mohammad, Wolach Baruch, Blancas-Galicia Lizbeth, Kanegane Hirokazu, Kawai Toshinao, Condino-Neto Antonio, Vihinen Mauno, Zerbe Christa S, Holland Steven M, Malech Harry L, Gallin John I, Kuhns Douglas B

СтатьяResearch Support, N.I.H., IntramuralПоддержка исследования: вне правительства СШАПеревод пока не подготовлен

Аннотация

Chronic granulomatous disease (CGD) is an immunodeficiency disorder affecting about 1 in 250,000 individuals. CGD patients suffer from severe, recurrent bacterial and fungal infections. The disease is caused by mutations in the genes encoding the components of the leukocyte NADPH oxidase. This enzyme produces superoxide, which is subsequently metabolized to hydrogen peroxide and other reactive oxygen species (ROS). These products are essential for intracellular killing of pathogens by phagocytic leukocytes (neutrophils, eosinophils, monocytes and macrophages). The leukocyte NADPH oxidase is composed of five subunits, four of which are encoded by autosomal genes. These are CYBA, encoding p22phox, NCF1, encoding p47phox, NCF2, encoding p67phox and NCF4, encoding p40phox. This article lists all mutations identified in these genes in CGD patients. In addition, cytochrome b558 chaperone-1 (CYBC1), recently recognized as an essential chaperone protein for the expression of the X-linked NADPH oxidase component gp91phox (also called Nox2), is encoded by the autosomal gene CYBC1. Mutations in this gene also lead to CGD. Finally, RAC2, a small GTPase of the Rho family, is needed for activation of the NADPH oxidase, and mutations in the RAC2 gene therefore also induce CGD-like symptoms. Mutations in these last two genes are also listed in this article.

Для цитирования: Roos Dirk, van Leeuwen Karin, Hsu Amy P, Priel Debra Long, Begtrup Amber, Brandon Rhonda, Rawat Amit, Vignesh Pandiarajan, Madkaikar Manesha, Stasia Marie José, Bakri Faris Ghalib, de Boer Martin, Roesler Joachim, Köker Nezihe, Köker M Yavuz, Jakobsen Marianne, Bustamante Jacinta, Garcia-Morato Maria Bravo, Shephard Juan Luis Valdivieso, Cagdas Deniz, Tezcan Ilhan, Sherkat Roya, Mortaz Esmaeil, Fayezi Abbas, Shahrooei Mohammad, Wolach Baruch, Blancas-Galicia Lizbeth, Kanegane Hirokazu, Kawai Toshinao, Condino-Neto Antonio, Vihinen Mauno, Zerbe Christa S, Holland Steven M, Malech Harry L, Gallin John I, Kuhns Douglas B. Hematologically important mutations: The autosomal forms of chronic granulomatous disease (third update).. Blood cells, molecules & diseases. 2021. DOI: 10.1016/j.bcmd.2021.102596.

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