Splice acceptor site mutation of the transporter associated with antigen processing-1 gene in human bare lymphocyte syndrome

被引:49
作者
Furukawa, H
Murata, S
Yabe, T
Shimbara, N
Keicho, N
Kashiwase, K
Watanabe, K
Ishikawa, Y
Akaza, T
Tadokoro, K
Tohma, S
Inoue, T
Tokunaga, K
Yamamoto, K
Tanaka, K
Juji, T
机构
[1] Japanese Red Cross Cent Blood Ctr, Dept Res, Shibuya Ku, Tokyo 1500012, Japan
[2] Univ Tokyo, Fac Med, Dept Med, Tokyo 113, Japan
[3] Tokyo Metropolitan Inst Med Sci, Tokyo 1138613, Japan
[4] Japan Sci & Technol Corp, CREST, Tokyo 1138613, Japan
[5] Sumitomo Elect Ind Ltd, Biomed Res & Dev Dept, Yokohama, Kanagawa 2448588, Japan
[6] Univ Tokyo, Grad Sch Med, Dept Human Genet, Tokyo 113, Japan
关键词
D O I
10.1172/JCI5335
中图分类号
R-3 [医学研究方法]; R3 [基础医学];
学科分类号
1001 ;
摘要
Expression of histocompatibility leukocyte antigen (HLA) class I molecules on the cell surface depends on the heterodimer of the transporter associated with antigen processing 1 and 2 (TAP1 and TAP2), which transport peptides cleaved by proteasome to the class I molecules. Defects in the TAP2 protein have been reported in two families with HLA class I deficiency, the so-called bare lymphocyte syndrome (BLS) type I. We have, to our knowledge, identified for the first time a splice site mutation in the TAP1 gene of another BLS patient. In addition, class I heavy chains (HCs) did not form the normal complex with tapasin in the endoplasmic reticulum (ER) of the cells of our patient.
引用
收藏
页码:755 / 758
页数:4
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