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SLC4A2、SLC26A7 和 SLC26A9 的改变导致胃神经内分泌肿瘤的酸碱失衡,并揭示了一种同时发生的多自身免疫情况的新机制。

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.

机构信息

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

Network of Research on Rare Diseases (CIBERER), 28029 Madrid, Spain.

出版信息

Cells. 2021 Dec 10;10(12):3500. doi: 10.3390/cells10123500.

Abstract

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.

摘要

自身免疫性多内分泌腺综合征 (APS) 被认为涉及免疫系统功能障碍,并涉及同一患者不同组织中同时发生的几种自身免疫性疾病;然而,由于其遗传基础和发病机制尚不清楚,因此它的准确诊断仍然是一个难题。我们之前的研究揭示了 ATP 酶 H+/K+ 转运亚基α(ATP4A)质子泵的改变,导致细胞内酸碱平衡失衡,为萎缩性胃炎和胃神经内分泌肿瘤伴继发性自身免疫性疾病提供了自身免疫的发病机制。在这里,我们提出了涉及胃疾病的 APS 的遗传探索,以了解多自身免疫发病机制的潜在发病机制。对五个自身免疫性甲状腺胃家族的全外显子组测序 (WES) 研究揭示了 SLC4A2、SLC26A7 和 SLC26A9 中的不同致病性变异,这些基因与 ATP4A 共同转运。探索性的体外研究表明,所发现的基因参与了一种基于酸碱平衡改变的发病机制。因此,我们基于提出的机制构建了一个包含 12 个基因的定制基因panel,以评估新的 69 名 APS 患者系列。在总共研究的患者中,发现了该panel 中的 12 个基因中有 64 个经过滤的潜在有害变异,而在健康对照组中则没有。我们的研究揭示了一组溶质载体,它们在受不同自身免疫性疾病影响的组织中共表达,为同时发生的病理提出了独特的遗传起源。这些结果确定了一种基于遗传的新型多自身免疫机制,不仅可以解释胃部疾病,还可以解释 APS 中的甲状腺胃病理和疾病同时发生,与临床偶然发现不同。这为预测和诊断同时发生的自身免疫性疾病以及患者的临床管理开辟了一个新窗口。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/884b/8700745/6b9743f6d769/cells-10-03500-g001.jpg

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