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线粒体 T10432C 突变对 Cika 牛的影响:Leber 遗传性视神经病变的“潜在”模型。

The Consequences of Mitochondrial T10432C Mutation in Cika Cattle: A "Potential" Model for Leber's Hereditary Optic Neuropathy.

机构信息

Department of Pathology, Croatian Veterinary Institute, 10000 Zagreb, Croatia.

Department of Animal Science, Faculty of Agriculture, University of Zagreb, 10000 Zagreb, Croatia.

出版信息

Int J Mol Sci. 2022 Jun 6;23(11):6335. doi: 10.3390/ijms23116335.

Abstract

While mitogenome mutations leading to pathological manifestations are rare, more than 200 such mutations have been described in humans. In contrast, pathogenic mitogenome mutations are rare in domestic animals and have not been described at all in cattle. In the small local Slovenian cattle breed Cika, we identified (next-generation sequencing) two cows with the T10432C mitogenome mutation in the ND4L gene, which corresponds to the human T10663C mutation known to cause Leber's hereditary optic neuropathy (LHON). Pedigree analysis revealed that the cows in which the mutation was identified belong to two different maternal lineages with 217 individual cows born between 1997 and 2020. The identified mutation and its maternal inheritance were confirmed by Sanger sequencing across multiple generations, whereas no single analysis revealed evidence of heteroplasmy. A closer clinical examination of one cow with the T10432C mutation revealed exophthalmos, whereas histopathological examination revealed retinal ablations, subretinal oedema, and haemorrhage. The results of these analyses confirm the presence of mitochondrial mutation T10432C with homoplasmic maternal inheritance as well as clinical and histopathological signs similar to LHON in humans. Live animals with the mutation could be used as a suitable animal model that can improve our understanding of the pathogenesis of LHON and other mitochondriopathies.

摘要

虽然导致病理性表现的线粒体基因突变很少见,但已经在人类中描述了超过 200 种这样的突变。相比之下,致病性线粒体基因突变在家畜中很少见,在牛中根本没有描述。在斯洛文尼亚的小型本地 Cika 牛品种中,我们通过(下一代测序)在 ND4L 基因中发现了两只 T10432C 线粒体突变的奶牛,这与已知导致 Leber 遗传性视神经病变(LHON)的人类 T10663C 突变相对应。系谱分析显示,发现突变的奶牛属于两个不同的母系,1997 年至 2020 年间出生了 217 头奶牛。通过对多个世代进行 Sanger 测序,确认了所鉴定的突变及其母系遗传,而没有单一分析显示异质性的证据。对携带 T10432C 突变的一只奶牛进行更密切的临床检查发现了眼球突出,而组织病理学检查则发现视网膜消融、视网膜下水肿和出血。这些分析的结果证实了具有纯合母系遗传的线粒体突变 T10432C 的存在,以及与人类 LHON 相似的临床和组织病理学迹象。携带该突变的活体动物可用作合适的动物模型,有助于我们更好地理解 LHON 和其他线粒体疾病的发病机制。

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