Pathogenic Interleukin-10 Receptor Alpha Variants in Humans - Balancing Natural Selection and Clinical Implications.


Journal

Journal of clinical immunology
ISSN: 1573-2592
Titre abrégé: J Clin Immunol
Pays: Netherlands
ID NLM: 8102137

Informations de publication

Date de publication:
02 2023
Historique:
received: 28 12 2021
accepted: 09 09 2022
pubmed: 13 11 2022
medline: 4 2 2023
entrez: 12 11 2022
Statut: ppublish

Résumé

Balancing natural selection is a process by which genetic variants arise in populations that are beneficial to heterozygous carriers, but pathogenic when homozygous. We systematically investigated the prevalence, structural, and functional consequences of pathogenic IL10RA variants that are associated with monogenic inflammatory bowel disease. We identify 36 non-synonymous and non-sense variants in the IL10RA gene. Since the majority of these IL10RA variants have not been functionally characterized, we performed a systematic screening of their impact on STAT3 phosphorylation upon IL-10 stimulation. Based on the geographic accumulation of confirmed pathogenic IL10RA variants in East Asia and in Northeast China, the distribution of infectious disorders worldwide, and the functional evidence of IL-10 signaling in the pathogenesis, we identify Schistosoma japonicum infection as plausible selection pressure driving variation in IL10RA. Consistent with this is a partially augmented IL-10 response in peripheral blood mononuclear cells from heterozygous variant carriers. A parasite-driven heterozygote advantage through reduced IL-10 signaling has implications for health care utilization in regions with high allele frequencies and potentially indicates pathogen eradication strategies that target IL-10 signaling.

Identifiants

pubmed: 36370291
doi: 10.1007/s10875-022-01366-7
pii: 10.1007/s10875-022-01366-7
pmc: PMC9892166
doi:

Substances chimiques

Receptors, Interleukin-10 0
Interleukin-10 130068-27-8
Interleukin-10 Receptor alpha Subunit 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

495-511

Subventions

Organisme : Wellcome Trust
ID : 106169/ZZ14/Z
Pays : United Kingdom

Informations de copyright

© 2022. The Author(s).

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Auteurs

Dominik Aschenbrenner (D)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK.
Novartis Institutes for BioMedical Research, Novartis Pharma AG, Basel, Switzerland.

Ziqing Ye (Z)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK.
Department of Gastroenterology, National Children's Medical Center, Children's Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102, China.

Ying Zhou (Y)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK.
Department of Gastroenterology, National Children's Medical Center, Children's Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102, China.

Wenhui Hu (W)

Department of Gastroenterology, National Children's Medical Center, Children's Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102, China.

Isabel Brooks (I)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK.

Isabelle Williams (I)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK.

Melania Capitani (M)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK.
SenTcell Ltd., London, UK.

Lisa Gartner (L)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK.

Daniel Kotlarz (D)

Dr. von Hauner Children's Hospital, Department of Pediatrics, University Hospital, Ludwig-Maximilians-Universität Munich, Munich, Germany.
Institute of Translational Genomics, Helmholtz Zentrum München - German Research Center for Environmental Health, Neuherberg, Germany.

Scott B Snapper (SB)

Boston Children's Hospital and Harvard Medical School, Boston, MA, 02115, USA.

Christoph Klein (C)

Dr. von Hauner Children's Hospital, Department of Pediatrics, University Hospital, Ludwig-Maximilians-Universität Munich, Munich, Germany.
Gene Center, LMU Munich, Munich, Germany.
Deutsche Zentrum für Infektionsforschung (DZIF) and Deutsches Zentrum für Kinder- und Jugendgesundheit, Partner site Munich, Munich, Germany.

Aleixo M Muise (AM)

SickKids Inflammatory Bowel Disease Centre and Cell Biology Program, Research Institute, The Hospital for Sick Children, Toronto, Canada.
Division of Gastroenterology, Hepatology, and Nutrition, Department of Pediatrics, Toronto, Canada.
The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada.

Brian D Marsden (BD)

Centre of Medicines Discovery, NDM, University of Oxford, Oxford, OX3 7DQ, UK.
Kennedy Institute of Rheumatology, NDORMS, University of Oxford, Oxford, OX3 7FY, UK.

Ying Huang (Y)

Department of Gastroenterology, National Children's Medical Center, Children's Hospital of Fudan University, 399 Wanyuan Road, Shanghai, 201102, China. yhuang815@163.com.

Holm H Uhlig (HH)

Translational Gastroenterology Unit, Experimental Medicine, John Radcliffe Hospital, University of Oxford, Oxford, OX3 9DU, UK. holm.uhlig@ndm.ox.ac.uk.
Department of Pediatrics, University of Oxford, Oxford, UK. holm.uhlig@ndm.ox.ac.uk.
Biomedical Research Center, University of Oxford, Oxford, UK. holm.uhlig@ndm.ox.ac.uk.

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