A novel mutation in RFXANK gene and low B cell count in a patient with MHC class II deficiency: a case report

被引:4
|
作者
Abolnezhadian, Farhad [1 ]
Dehghani, Razieh [1 ]
Dehnavi, Sajad [2 ,3 ]
Khodadadi, Ali [2 ]
Shohan, Mojtaba [2 ,3 ]
机构
[1] Ahvaz Jundishapur Univ Med Sci, Abuzar Childrens Hosp, Dept Pediat, Ahvaz, Iran
[2] Ahvaz Jundishapur Univ Med Sci, Dept Immunol, Fac Med, Ahvaz, Iran
[3] Ahvaz Jundishapur Univ Med Sci, Student Res Comm, Ahvaz, Iran
关键词
MHC class II deficiency; RFXANK; Primary immunodeficiency disorder; Whole exome sequencing; PRIMARY IMMUNODEFICIENCY DISEASES; BARE LYMPHOCYTE SYNDROME; DIAGNOSIS;
D O I
10.1007/s12026-020-09141-9
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
Recurrence of severe microbial infections results from a primary immunodeficiency disorder known as major histocompatibility complex class II deficiency or bare lymphocyte syndrome type II. Immunologic function is severely impaired due to the absence of MHC class II molecules on the surface of immune cells. Here, we report a 5-year-old boy with a novel homozygous mutation in RFXANK gene that negatively affects the proper expression of MHC class II molecules by antigen presenting cells. The frame shift mutations in RFXANK gene and negative HLA-DR proteins expression on peripheral blood mononuclear cells were identified and confirmed by whole exome sequencing, Sanger sequencing, and flow cytometry. The patient was referred with long-term severe prolonged diarrhea, fever, coughing, and vomiting. Also, antibiotic resistance, normal T cell, and NK cell counts with low B cell count and reduced serum immunoglobulin level were observed. The patient did not give a dramatic response to intravenous immunoglobulin infusion. The significancy of this report is the novelty of mutation and low B cell count which is not commonly expected in such patients. The final diagnosis of BLS type II is based on WES, Sanger sequencing, and flow cytometric evaluation. Moreover, it seems that the only therapeutic choice is hematopoietic stem cell transplantation.
引用
收藏
页码:225 / 231
页数:7
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