Impact of rare and common genetic variation in the interleukin-1 pathway on human cytokine responses.


Journal

Genome medicine
ISSN: 1756-994X
Titre abrégé: Genome Med
Pays: England
ID NLM: 101475844

Informations de publication

Date de publication:
25 05 2021
Historique:
received: 04 12 2020
accepted: 11 05 2021
entrez: 26 5 2021
pubmed: 27 5 2021
medline: 19 2 2022
Statut: epublish

Résumé

The interleukin (IL)-1 pathway is primarily associated with innate immunological defense and plays a major role in the induction and regulation of inflammation. Both common and rare genetic variation in this pathway underlies various inflammation-mediated diseases, but the role of rare variants relative to common variants in immune response variability in healthy individuals remains unclear. We performed molecular inversion probe sequencing on 48 IL-1 pathway-related genes in 463 healthy individuals from the Human Functional Genomics Project. We functionally grouped common and rare variants, over gene, subpathway, and inflammatory levels and performed the Sequence Kernel Association Test to test for association with in vitro stimulation-induced cytokine responses; specifically, IL-1β and IL-6 cytokine measurements upon stimulations that represent an array of microbial infections: lipopolysaccharide (LPS), phytohaemagglutinin (PHA), Candida albicans (C. albicans), and Staphylococcus aureus (S. aureus). We identified a burden of NCF4 rare variants with PHA-induced IL-6 cytokine and showed that the respective carriers are in the 1% lowest IL-6 producers. Collapsing rare variants in IL-1 subpathway genes produces a bidirectional association with LPS-induced IL-1β cytokine levels, which is reflected by a significant Spearman correlation. On the inflammatory level, we identified a burden of rare variants in genes encoding for proteins with an anti-inflammatory function with S. aureus-induced IL-6 cytokine. In contrast to these rare variant findings which were based on different types of stimuli, common variant associations were exclusively identified with C. albicans-induced cytokine over various levels of grouping, from the gene, to subpathway, to inflammatory level. In conclusion, this study shows that functionally grouping common and rare genetic variants enables the elucidation IL-1-mediated biological mechanisms, specifically, for IL-1β and IL-6 cytokine responses induced by various stimuli. The framework used in this study may allow for the analysis of rare and common genetic variants in a wider variety of (non-immune) complex phenotypes and therefore has the potential to contribute to better understanding of unresolved, complex traits and diseases.

Sections du résumé

BACKGROUND
The interleukin (IL)-1 pathway is primarily associated with innate immunological defense and plays a major role in the induction and regulation of inflammation. Both common and rare genetic variation in this pathway underlies various inflammation-mediated diseases, but the role of rare variants relative to common variants in immune response variability in healthy individuals remains unclear.
METHODS
We performed molecular inversion probe sequencing on 48 IL-1 pathway-related genes in 463 healthy individuals from the Human Functional Genomics Project. We functionally grouped common and rare variants, over gene, subpathway, and inflammatory levels and performed the Sequence Kernel Association Test to test for association with in vitro stimulation-induced cytokine responses; specifically, IL-1β and IL-6 cytokine measurements upon stimulations that represent an array of microbial infections: lipopolysaccharide (LPS), phytohaemagglutinin (PHA), Candida albicans (C. albicans), and Staphylococcus aureus (S. aureus).
RESULTS
We identified a burden of NCF4 rare variants with PHA-induced IL-6 cytokine and showed that the respective carriers are in the 1% lowest IL-6 producers. Collapsing rare variants in IL-1 subpathway genes produces a bidirectional association with LPS-induced IL-1β cytokine levels, which is reflected by a significant Spearman correlation. On the inflammatory level, we identified a burden of rare variants in genes encoding for proteins with an anti-inflammatory function with S. aureus-induced IL-6 cytokine. In contrast to these rare variant findings which were based on different types of stimuli, common variant associations were exclusively identified with C. albicans-induced cytokine over various levels of grouping, from the gene, to subpathway, to inflammatory level.
CONCLUSIONS
In conclusion, this study shows that functionally grouping common and rare genetic variants enables the elucidation IL-1-mediated biological mechanisms, specifically, for IL-1β and IL-6 cytokine responses induced by various stimuli. The framework used in this study may allow for the analysis of rare and common genetic variants in a wider variety of (non-immune) complex phenotypes and therefore has the potential to contribute to better understanding of unresolved, complex traits and diseases.

Identifiants

pubmed: 34034819
doi: 10.1186/s13073-021-00907-w
pii: 10.1186/s13073-021-00907-w
pmc: PMC8145796
doi:

Substances chimiques

Biomarkers 0
Cytokines 0
Interleukin-1 0
Interleukin-1beta 0

Types de publication

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

Langues

eng

Sous-ensembles de citation

IM

Pagination

94

Subventions

Organisme : NIAID NIH HHS
ID : R01 AI015614
Pays : United States

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Auteurs

Rosanne C van Deuren (RC)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands.

Peer Arts (P)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Genetics and Molecular Pathology, Centre for Cancer Biology, SA Pathology and the University of South Australia, Adelaide, South Australia, Australia.

Giulio Cavalli (G)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Unit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Hospital and Vita-Salute San Raffaele University, Milan, Italy.
Department of Medicine, University of Colorado, Aurora, CO, USA.

Martin Jaeger (M)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands.

Marloes Steehouwer (M)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.

Maartje van de Vorst (M)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.

Christian Gilissen (C)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands.

Leo A B Joosten (LAB)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Medical Genetics, Iuliu Hatieganu University of Medicine and Pharmacy, Cluj-Napoca, Romania.

Charles A Dinarello (CA)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Medicine, University of Colorado, Aurora, CO, USA.

Musa M Mhlanga (MM)

Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands.
Epigenomics & Single Cell Biophysics Group, Department of Cell Biology, Radboud University, 6525 GA, Nijmegen, The Netherlands.

Vinod Kumar (V)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Department of Genetics, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands.
Nitte (Deemed to be University), Nitte University Centre for Science Education and Research (NUCSER), Medical Sciences Complex, Deralakatte, Mangalore, 575018, India.

Mihai G Netea (MG)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands.
Department for Genomics & Immunoregulation, Life and Medical Sciences Institute (LIMES), University of Bonn, Bonn, Germany.

Frank L van de Veerdonk (FL)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands.

Alexander Hoischen (A)

Department of Internal Medicine, Radboud Expertise Center for Immunodeficiency and Autoinflammation, and Radboud Center for Infectious Disease (RCI), Radboud University Medical Center, Nijmegen, the Netherlands. Alexander.Hoischen@radboudumc.nl.
Department of Human Genetics, Radboud University Medical Center, Nijmegen, the Netherlands. Alexander.Hoischen@radboudumc.nl.
Radboud Institute of Molecular Life Sciences (RIMLS), Radboud University Medical Center, Nijmegen, the Netherlands. Alexander.Hoischen@radboudumc.nl.

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