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Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders?
Primary immunodeficiency (PID) disorders, also commonly referred to as inborn errors of immunity, are a heterogenous group of human genetic diseases characterized by defects in immune cell development and/or function. Since these disorders are generally uncommon and occur on a variable background pr...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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MDPI
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10095346/ https://www.ncbi.nlm.nih.gov/pubmed/37047441 http://dx.doi.org/10.3390/ijms24076468 |
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author | Basheer, Faiza Sertori, Robert Liongue, Clifford Ward, Alister C. |
author_facet | Basheer, Faiza Sertori, Robert Liongue, Clifford Ward, Alister C. |
author_sort | Basheer, Faiza |
collection | PubMed |
description | Primary immunodeficiency (PID) disorders, also commonly referred to as inborn errors of immunity, are a heterogenous group of human genetic diseases characterized by defects in immune cell development and/or function. Since these disorders are generally uncommon and occur on a variable background profile of potential genetic and environmental modifiers, animal models are critical to provide mechanistic insights as well as to create platforms to underpin therapeutic development. This review aims to review the relevance of zebrafish as an alternative genetic model for PIDs. It provides an overview of the conservation of the zebrafish immune system and details specific examples of zebrafish models for a multitude of specific human PIDs across a range of distinct categories, including severe combined immunodeficiency (SCID), combined immunodeficiency (CID), multi-system immunodeficiency, autoinflammatory disorders, neutropenia and defects in leucocyte mobility and respiratory burst. It also describes some of the diverse applications of these models, particularly in the fields of microbiology, immunology, regenerative biology and oncology. |
format | Online Article Text |
id | pubmed-10095346 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-100953462023-04-13 Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders? Basheer, Faiza Sertori, Robert Liongue, Clifford Ward, Alister C. Int J Mol Sci Review Primary immunodeficiency (PID) disorders, also commonly referred to as inborn errors of immunity, are a heterogenous group of human genetic diseases characterized by defects in immune cell development and/or function. Since these disorders are generally uncommon and occur on a variable background profile of potential genetic and environmental modifiers, animal models are critical to provide mechanistic insights as well as to create platforms to underpin therapeutic development. This review aims to review the relevance of zebrafish as an alternative genetic model for PIDs. It provides an overview of the conservation of the zebrafish immune system and details specific examples of zebrafish models for a multitude of specific human PIDs across a range of distinct categories, including severe combined immunodeficiency (SCID), combined immunodeficiency (CID), multi-system immunodeficiency, autoinflammatory disorders, neutropenia and defects in leucocyte mobility and respiratory burst. It also describes some of the diverse applications of these models, particularly in the fields of microbiology, immunology, regenerative biology and oncology. MDPI 2023-03-30 /pmc/articles/PMC10095346/ /pubmed/37047441 http://dx.doi.org/10.3390/ijms24076468 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Review Basheer, Faiza Sertori, Robert Liongue, Clifford Ward, Alister C. Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders? |
title | Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders? |
title_full | Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders? |
title_fullStr | Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders? |
title_full_unstemmed | Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders? |
title_short | Zebrafish: A Relevant Genetic Model for Human Primary Immunodeficiency (PID) Disorders? |
title_sort | zebrafish: a relevant genetic model for human primary immunodeficiency (pid) disorders? |
topic | Review |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10095346/ https://www.ncbi.nlm.nih.gov/pubmed/37047441 http://dx.doi.org/10.3390/ijms24076468 |
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