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Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation
Polyglandular autoimmune syndrome type 1, also known as autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED), is a rare primary immunodeficiency disorder with multi-organ involvement. Besides for being predisposed to severe life-threatening infections, patients with APECED are als...
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Hindawi
2019
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Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6949663/ https://www.ncbi.nlm.nih.gov/pubmed/31956453 http://dx.doi.org/10.1155/2019/8746249 |
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author | Gavrilova, Tatyana |
author_facet | Gavrilova, Tatyana |
author_sort | Gavrilova, Tatyana |
collection | PubMed |
description | Polyglandular autoimmune syndrome type 1, also known as autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED), is a rare primary immunodeficiency disorder with multi-organ involvement. Besides for being predisposed to severe life-threatening infections, patients with APECED are also prone to organ impairment secondary to severe autoimmunity. As this is an autosomal recessive disorder, a biallelic mutation in the AIRE gene is responsible for APECED. The author presents a case of APECED with a single AIRE mutation. Whole exome sequencing identified a mutation in the BTNL2 gene that the author suggests may have contributed to the patient's presentation. |
format | Online Article Text |
id | pubmed-6949663 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2019 |
publisher | Hindawi |
record_format | MEDLINE/PubMed |
spelling | pubmed-69496632020-01-17 Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation Gavrilova, Tatyana Case Reports Immunol Case Report Polyglandular autoimmune syndrome type 1, also known as autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED), is a rare primary immunodeficiency disorder with multi-organ involvement. Besides for being predisposed to severe life-threatening infections, patients with APECED are also prone to organ impairment secondary to severe autoimmunity. As this is an autosomal recessive disorder, a biallelic mutation in the AIRE gene is responsible for APECED. The author presents a case of APECED with a single AIRE mutation. Whole exome sequencing identified a mutation in the BTNL2 gene that the author suggests may have contributed to the patient's presentation. Hindawi 2019-12-28 /pmc/articles/PMC6949663/ /pubmed/31956453 http://dx.doi.org/10.1155/2019/8746249 Text en Copyright © 2019 Tatyana Gavrilova. http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. |
spellingShingle | Case Report Gavrilova, Tatyana Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation |
title | Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation |
title_full | Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation |
title_fullStr | Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation |
title_full_unstemmed | Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation |
title_short | Primary Immunodeficiency with Severe Multi-Organ Immune Dysregulation |
title_sort | primary immunodeficiency with severe multi-organ immune dysregulation |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6949663/ https://www.ncbi.nlm.nih.gov/pubmed/31956453 http://dx.doi.org/10.1155/2019/8746249 |
work_keys_str_mv | AT gavrilovatatyana primaryimmunodeficiencywithseveremultiorganimmunedysregulation |