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Erschienen in: Immunologic Research 4/2020

23.06.2020 | Brief report

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

verfasst von: Farhad Abolnezhadian, Razieh Dehghani, Sajad Dehnavi, Ali Khodadadi, Mojtaba Shohan

Erschienen in: Immunologic Research | Ausgabe 4/2020

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Abstract

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.
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Metadaten
Titel
A novel mutation in RFXANK gene and low B cell count in a patient with MHC class II deficiency: a case report
verfasst von
Farhad Abolnezhadian
Razieh Dehghani
Sajad Dehnavi
Ali Khodadadi
Mojtaba Shohan
Publikationsdatum
23.06.2020
Verlag
Springer US
Erschienen in
Immunologic Research / Ausgabe 4/2020
Print ISSN: 0257-277X
Elektronische ISSN: 1559-0755
DOI
https://doi.org/10.1007/s12026-020-09141-9

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