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DOI 10.1534/g3.117.039511

Медицинская библиотека · G3 (Bethesda, Md.) · 2017

A Nonsense Variant in the ST14 Gene in Akhal-Teke Horses with Naked Foal Syndrome.

Bauer Anina, Hiemesch Theresa, Jagannathan Vidhya, Neuditschko Markus, Bachmann Iris, Rieder Stefan, Mikko Sofia, Penedo M Cecilia, Tarasova Nadja, Vitková Martina, Sirtori Nicolò, Roccabianca Paola, Leeb Tosso, Welle Monika M

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Аннотация

Naked foal syndrome (NFS) is a genodermatosis in the Akhal-Teke horse breed. We provide the first scientific description of this phenotype. Affected horses have almost no hair and show a mild ichthyosis. So far, all known NFS affected horses died between a few weeks and 3 yr of age. It is not clear whether a specific pathology caused the premature deaths. NFS is inherited as a monogenic autosomal recessive trait. We mapped the disease causing genetic variant to two segments on chromosomes 7 and 27 in the equine genome. Whole genome sequencing of two affected horses, two obligate carriers, and 75 control horses from other breeds revealed a single nonsynonymous genetic variant on the chromosome 7 segment that was perfectly associated with NFS. The affected horses were homozygous for ST14:c.388G>T, a nonsense variant that truncates >80% of the open reading frame of the ST14 gene (p.Glu130*). The variant leads to partial nonsense-mediated decay of the mutant transcript. Genetic variants in the ST14 gene are responsible for autosomal recessive congenital ichthyosis 11 in humans. Thus, the identified equine ST14:c.388G>T variant is an excellent candidate causative variant for NFS, and the affected horses represent a large animal model for a known human genodermatosis. Our findings will enable genetic testing to avoid the nonintentional breeding of NFS-affected foals.

Для цитирования: Bauer Anina, Hiemesch Theresa, Jagannathan Vidhya, Neuditschko Markus, Bachmann Iris, Rieder Stefan, Mikko Sofia, Penedo M Cecilia, Tarasova Nadja, Vitková Martina, Sirtori Nicolò, Roccabianca Paola, Leeb Tosso, Welle Monika M. A Nonsense Variant in the ST14 Gene in Akhal-Teke Horses with Naked Foal Syndrome.. G3 (Bethesda, Md.). 2017. DOI: 10.1534/g3.117.039511.

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