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.
achlorhydria
autoimmune polyendocrine syndrome (APS)
autoimmune thyrogastric syndrome
gastric neuroendocrine tumors (gNETS)
immunodeficiencies
solute carriers (SLCs)
Journal
Cells
ISSN: 2073-4409
Titre abrégé: Cells
Pays: Switzerland
ID NLM: 101600052
Informations de publication
Date de publication:
10 12 2021
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
Références
Gastric Cancer. 2017 Nov;20(6):998-1003
pubmed: 28474257
BMC Med. 2018 Feb 20;16(1):25
pubmed: 29458357
Autoimmun Rev. 2014 Feb;13(2):85-9
pubmed: 24055063
Cancers (Basel). 2020 Mar 13;12(3):
pubmed: 32183222
N Engl J Med. 2018 Mar 22;378(12):1132-1141
pubmed: 29562162
J Autoimmun. 1995 Feb;8(1):121-30
pubmed: 7734032
Dermatol Clin. 2017 Apr;35(2):245-255
pubmed: 28317533
J Clin Endocrinol Metab. 2000 Feb;85(2):688-91
pubmed: 10690877
Gastric Cancer. 2020 Jan;23(1):52-63
pubmed: 31250150
Hum Mol Genet. 2015 May 15;24(10):2914-22
pubmed: 25678551
Endocr Rev. 2002 Jun;23(3):327-64
pubmed: 12050123
J Biol Chem. 2009 Oct 23;284(43):29470-9
pubmed: 19723628
Rev Med Liege. 2013 Nov;68(11):579-84
pubmed: 24396972
Nat Rev Gastroenterol Hepatol. 2013 Sep;10(9):529-41
pubmed: 23774773
Autoimmun Rev. 2019 Mar;18(3):215-222
pubmed: 30639639
J Clin Endocrinol Metab. 2008 Feb;93(2):363-71
pubmed: 18029461
Nat Rev Rheumatol. 2017 Dec 19;14(1):7-18
pubmed: 29255211
Nat Immunol. 2017 Jul 19;18(8):832-842
pubmed: 28722725
J Allergy Clin Immunol. 2017 Nov;140(5):1388-1393.e8
pubmed: 28192146
Nat Rev Immunol. 2016 Apr;16(4):247-58
pubmed: 26972725
J Clin Endocrinol Metab. 2018 May 1;103(5):1889-1898
pubmed: 29546359
Clin Exp Gastroenterol. 2017 Feb 07;10:19-27
pubmed: 28223833
Dis Model Mech. 2016 Sep 1;9(9):975-84
pubmed: 27491072
Rheumatology (Oxford). 2019 Nov 1;58(Suppl 6):vi1-vi8
pubmed: 31769855
Front Med (Lausanne). 2021 Apr 06;8:553110
pubmed: 33889580
J Hepatol. 2009 Mar;50(3):633-5
pubmed: 19157624
Autoimmun Rev. 2015 Feb;14(2):174-80
pubmed: 25461470
Autoimmun Rev. 2011 Jun;10(8):455-9
pubmed: 21296191
Proc Natl Acad Sci U S A. 2008 Nov 18;105(46):17955-60
pubmed: 19004773
Diabetes Care. 2016 Jun;39(6):893-901
pubmed: 27222547
Compr Physiol. 2019 Mar 15;9(2):839-872
pubmed: 30873581
JCI Insight. 2018 Oct 18;3(20):
pubmed: 30333321
J Formos Med Assoc. 2006 Nov;105(11):946-9
pubmed: 17098697
Clin Exp Dermatol. 2006 Sep;31(5):746-9
pubmed: 16803462
Front Endocrinol (Lausanne). 2017 Apr 26;8:92
pubmed: 28491051