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
相关论文
共 50 条
  • [31] Two MHC class II deficiency patients are characterized by novel mutations in the RFX5 gene
    Peijnenburg, A
    Van Eggermond, M
    Van den Berg, R
    Gobin, S
    Van den Elsen, P
    EUROPEAN JOURNAL OF HUMAN GENETICS, 1998, 6 : 71 - 71
  • [32] The 752delG26 mutation in the RFXANK gene associated with major histocompatibility complex class II deficiency: evidence for a founder effect in the Moroccan population
    Hamid Naamane
    Ouafaa El Maataoui
    Fatima Ailal
    Abdelhamid Barakat
    Siham Bennani
    Jilali Najib
    Mohammed Hassar
    Rachid Saile
    Ahmed Aziz Bousfiha
    European Journal of Pediatrics, 2010, 169 : 1069 - 1074
  • [33] MHC class II deficiency in the Dene native population: a case report highlighting pitfalls in diagnosis and treatment
    Alex Lyttle
    Chaim Roifman
    Harjit Dadi
    Nicola Wright
    Fotini Kavadas
    Allergy, Asthma & Clinical Immunology, 10 (Suppl 1)
  • [34] First case report of cerebral folate deficiency caused by a novel mutation of FOLR1 gene in a Chinese patient
    Zhang, Ciliu
    Deng, Xiaolu
    Wen, Yafei
    He, Fang
    Yin, Fei
    Peng, Jing
    BMC MEDICAL GENETICS, 2020, 21 (01)
  • [35] Case Report: A Novel Mutation Leading to 11-β Hydroxylase Deficiency in a Female Patient
    Ozbas, Burak
    Demir, Mikail
    Dursun, Huseyin
    Sahin, Izem
    Hacioglu, Aysa
    Karaca, Zuleyha
    Dundar, Munis
    Unluhizarci, Kursad
    ENDOCRINE METABOLIC & IMMUNE DISORDERS-DRUG TARGETS, 2023, 23 (05) : 721 - 726
  • [36] Case report: A Chinese patient with glutathione synthetase deficiency and a novel glutathione synthase mutation
    Wu, Xiaojiao
    Jiao, Jiancheng
    Xia, Yaofang
    Yan, Xiaotong
    Liu, Zehao
    Cao, Yanyan
    Ma, Li
    FRONTIERS IN PEDIATRICS, 2023, 11
  • [37] A gene encoding a novel RFX-associated transactivator is mutated in the majority of MHC class II deficiency patients
    Masternak, K
    Barras, E
    Zufferey, M
    Conrad, B
    Corthals, G
    Aebersold, R
    Sanchez, JC
    Hochstrasser, DF
    Mach, B
    Reith, W
    NATURE GENETICS, 1998, 20 (03) : 273 - 277
  • [38] A gene encoding a novel RFX-associated transactivator is mutated in the majority of MHC class II deficiency patients
    Krzysztof Masternak
    Emmanuèle Barras
    Madeleine Zufferey
    Bernard Conrad
    Garry Corthals
    Ruedi Aebersold
    Jean-Charles Sanchez
    Denis F. Hochstrasser
    Bernard Mach
    Walter Reith
    Nature Genetics, 1998, 20 : 273 - 277
  • [39] Congenital hypothyroidism due to thyroglobulin deficiency: a case report with a novel mutation in TG gene
    Heo, Seung
    Jang, Ja-Hyun
    Yu, Jeesuk
    ANNALS OF PEDIATRIC ENDOCRINOLOGY & METABOLISM, 2019, 24 (03) : 199 - 202
  • [40] Novel SOST gene mutation in a sclerosteosis patient from Morocco: A case report
    Belkhribchia, Mohamed Reda
    Collet, Corinne
    Laplanche, Jean-Louis
    Hassani, Redouane
    EUROPEAN JOURNAL OF MEDICAL GENETICS, 2014, 57 (04) : 133 - 137