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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...

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Detalles Bibliográficos
Autores principales: Basheer, Faiza, Sertori, Robert, Liongue, Clifford, Ward, Alister C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
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.
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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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