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.
机构:
Shiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, IranShiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
Mirzaei, Malihe
Kavosi, Arghavan
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机构:
Shiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, IranShiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
Kavosi, Arghavan
Sharifzadeh, Mahboobeh
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机构:
Islamic Azad Univ, Dept Biol, Arsanjan Branch, Arsanjan, IranShiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
Sharifzadeh, Mahboobeh
Mahjoub, Ghazale
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机构:
Shiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, IranShiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
Mahjoub, Ghazale
Faghihi, Mohammad Ali
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机构:
Shiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
Univ Miami, Miller Sch Med, Dept Psychiat & Behav Sci, Miami, FL 33136 USAShiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
Faghihi, Mohammad Ali
Habibzadeh, Parham
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机构:
Shiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
Shiraz Univ Med Sci, Student Res Comm, Shiraz, IranShiraz Univ Med Sci, Persian BayanGene Res & Training Ctr, Shiraz 7134767617, Iran
机构:
Sichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Sichuan Univ, Minist Educ, Key Lab Birth Defects & Related Dis Women & Childr, Chengdu, Sichuan, Peoples R ChinaSichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Tan, Yu
Tian, Huan
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机构:
Sichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Sichuan Univ, Minist Educ, Key Lab Birth Defects & Related Dis Women & Childr, Chengdu, Sichuan, Peoples R ChinaSichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Tian, Huan
Mai, Jingqun
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机构:
Sichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Sichuan Univ, Minist Educ, Key Lab Birth Defects & Related Dis Women & Childr, Chengdu, Sichuan, Peoples R ChinaSichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Mai, Jingqun
Yang, Shuo
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机构:
Sichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Sichuan Univ, Minist Educ, Key Lab Birth Defects & Related Dis Women & Childr, Chengdu, Sichuan, Peoples R ChinaSichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Yang, Shuo
Wang, He
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机构:
Sichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Sichuan Univ, Minist Educ, Key Lab Birth Defects & Related Dis Women & Childr, Chengdu, Sichuan, Peoples R ChinaSichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Wang, He
Yang, Mei
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机构:
Sichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Sichuan Univ, Minist Educ, Key Lab Birth Defects & Related Dis Women & Childr, Chengdu, Sichuan, Peoples R ChinaSichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Yang, Mei
Liu, Shanling
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机构:
Sichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
Sichuan Univ, Minist Educ, Key Lab Birth Defects & Related Dis Women & Childr, Chengdu, Sichuan, Peoples R ChinaSichuan Univ, West China Univ Hosp 2, Dept Med Genet, Chengdu, Sichuan, Peoples R China
机构:
Cheeloo College of Medicine,Shandong UniversityCheeloo College of Medicine,Shandong University
Xing-Chen Wang
Ting Wang
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机构:
Department of Neurology,Affiliated Hospital of Jining Medical UniversityCheeloo College of Medicine,Shandong University
Ting Wang
Rui-Han Liu
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h-index: 0
机构:
Department of Pediatrics,Affiliated Hospital of Jining Medical University
College of TCM,Shandong University of Traditional Chinese MedicineCheeloo College of Medicine,Shandong University
Rui-Han Liu
Yan Jiang
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机构:
Clinical Medical College,Jining Medical UniversityCheeloo College of Medicine,Shandong University
Yan Jiang
Dan-Dan Chen
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机构:
Clinical Medical College,Jining Medical UniversityCheeloo College of Medicine,Shandong University
Dan-Dan Chen
Xin-Yu Wang
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机构:
Clinical Medical College,Jining Medical UniversityCheeloo College of Medicine,Shandong University
Xin-Yu Wang
Qing-Xia Kong
论文数: 0引用数: 0
h-index: 0
机构:
Department of Neurology,Affiliated Hospital of Jining Medical UniversityCheeloo College of Medicine,Shandong University
机构:
Dalian Med Univ, Hosp 2, Dept Gastroenterol, Dalian, Peoples R ChinaDalian Med Univ, Hosp 2, Dept Gastroenterol, Dalian, Peoples R China
Tan, Shiqing
Wu, Xiaoting
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机构:
Dalian Med Univ, Hosp 2, Dept Dermatol, Dalian, Peoples R China
Zhejiang Chinese Med Univ, Clin Med Coll 3, Hangzhou, Peoples R ChinaDalian Med Univ, Hosp 2, Dept Gastroenterol, Dalian, Peoples R China
Wu, Xiaoting
Wang, Aoxue
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机构:
Dalian Med Univ, Hosp 2, Dept Dermatol, Dalian, Peoples R ChinaDalian Med Univ, Hosp 2, Dept Gastroenterol, Dalian, Peoples R China
Wang, Aoxue
Ying, Li
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机构:
Dalian Med Univ, Hosp 2, Dept Gastroenterol, Dalian, Peoples R ChinaDalian Med Univ, Hosp 2, Dept Gastroenterol, Dalian, Peoples R China