Alterations in SLC4A2, SLC26A7 and SLC26A9 Drive Acid-Base Imbalance in Gastric Neuroendocrine Tumors and Uncover a Novel Mechanism for a Co-Occurring Polyautoimmune Scenario.


Journal

Cells
ISSN: 2073-4409
Titre abrégé: Cells
Pays: Switzerland
ID NLM: 101600052

Informations de publication

Date de publication:
10 12 2021
Historique:
received: 23 11 2021
accepted: 07 12 2021
entrez: 24 12 2021
pubmed: 25 12 2021
medline: 6 1 2022
Statut: epublish

Résumé

Autoimmune polyendocrine syndrome (APS) is assumed to involve an immune system malfunction and entails several autoimmune diseases co-occurring in different tissues of the same patient; however, they are orphans of its accurate diagnosis, as its genetic basis and pathogenic mechanism are not understood. Our previous studies uncovered alterations in the ATPase H+/K+ Transporting Subunit Alpha (ATP4A) proton pump that triggered an internal cell acid-base imbalance, offering an autoimmune scenario for atrophic gastritis and gastric neuroendocrine tumors with secondary autoimmune pathologies. Here, we propose the genetic exploration of APS involving gastric disease to understand the underlying pathogenic mechanism of the polyautoimmune scenario. The whole exome sequencing (WES) study of five autoimmune thyrogastric families uncovered different pathogenic variants in SLC4A2, SLC26A7 and SLC26A9, which cotransport together with ATP4A. Exploratory in vitro studies suggested that the uncovered genes were involved in a pathogenic mechanism based on the alteration of the acid-base balance. Thus, we built a custom gene panel with 12 genes based on the suggested mechanism to evaluate a new series of 69 APS patients. In total, 64 filtered putatively damaging variants in the 12 genes of the panel were found in 54.17% of the studied patients and none of the healthy controls. Our studies reveal a constellation of solute carriers that co-express in the tissues affected with different autoimmune diseases, proposing a unique genetic origin for co-occurring pathologies. These results settle a new-fangled genetics-based mechanism for polyautoimmunity that explains not only gastric disease, but also thyrogastric pathology and disease co-occurrence in APS that are different from clinical incidental findings. This opens a new window leading to the prediction and diagnosis of co-occurring autoimmune diseases and clinical management of patients.

Identifiants

pubmed: 34944008
pii: cells10123500
doi: 10.3390/cells10123500
pmc: PMC8700745
pii:
doi:

Substances chimiques

Antiporters 0
Chloride-Bicarbonate Antiporters 0
SLC26A7 protein, human 0
SLC26A9 protein, human 0
SLC4A2 protein, human 0
Sulfate Transporters 0

Types de publication

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

Langues

eng

Sous-ensembles de citation

IM

Subventions

Organisme : Instituto de Salud Carlos III
ID : PI16/00440
Organisme : H2020 BRIDGES project
ID : 634935

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Auteurs

Oriol Calvete (O)

Human Genetics Group, Spanish National Cancer Research Center (CNIO), 28029 Madrid, Spain.
Network of Research on Rare Diseases (CIBERER), 28029 Madrid, Spain.
Grupo Español de Tumores Neuroendocrinos y Endocrinos (GETNE), 28054 Madrid, Spain.

José Reyes (J)

Grupo Español de Tumores Neuroendocrinos y Endocrinos (GETNE), 28054 Madrid, Spain.
Department of Gastroenterology, Hospital Comarcal de Inca, 07300 Inca, Spain.
Health Investigation Institute (IDISBA), 07120 Palma de Mallorca, Spain.

Hernán Valdés-Socin (H)

Department of Endocrinology, Centre Hospitalier Universitaire de Liège, 4000 Liège, Belgium.

Paloma Martin (P)

Human Genetics Group, Spanish National Cancer Research Center (CNIO), 28029 Madrid, Spain.
Network of Research on Rare Diseases (CIBERER), 28029 Madrid, Spain.

Mónica Marazuela (M)

Hospital la Princesa, Instituto de Investigación Princesa, University Autónoma of Madrid, 28006 Madrid, Spain.

Alicia Barroso (A)

Human Genetics Group, Spanish National Cancer Research Center (CNIO), 28029 Madrid, Spain.

Javier Escalada (J)

Endocrinology and Nutrition Department, Clínica Universidad de Navarra, 31008 Pamplona, Spain.
IdiSNA, Navarra Institute for Health Research, 31008 Pamplona, Spain.
CIBER Fisiopatología de la Obesidad y Nutrición (CIBEROBN), Instituto de Salud Carlos III, 28009 Madrid, Spain.

Antoni Castells (A)

Hospital Clinic of Barcelona, IDIBAPS, CIBEREHD, University of Barcelona, 08036 Barcelona, Spain.

Raúl Torres-Ruiz (R)

Molecular Cytogenetics and Genome Editing Unit, Spanish National Cancer Research Center (CNIO), 28029 Madrid, Spain.

Sandra Rodríguez-Perales (S)

Molecular Cytogenetics and Genome Editing Unit, Spanish National Cancer Research Center (CNIO), 28029 Madrid, Spain.

María Currás-Freixes (M)

Endocrinology and Nutrition Department, Clínica Universidad de Navarra, 28027 Madrid, Spain.

Javier Benítez (J)

Human Genetics Group, Spanish National Cancer Research Center (CNIO), 28029 Madrid, Spain.
Network of Research on Rare Diseases (CIBERER), 28029 Madrid, Spain.

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Classifications MeSH