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Genes (Basel) ; 12(10)2021 09 24.
Artículo en Inglés | MEDLINE | ID: mdl-34680883

RESUMEN

Dwarfism phenotypes occur in many species and may be caused by genetic or environmental factors. In this study, we investigated a family of nine Dogo Argentino dogs, in which two dogs were affected by disproportionate dwarfism. Radiographs of an affected dog revealed a decreased level of endochondral ossification in its growth plates, and a premature closure of the distal ulnar physes. The pedigree of the dogs presented evidence of monogenic autosomal recessive inheritance; combined linkage and homozygosity mapping assigned the most likely position of a potential genetic defect to 34 genome segments, totaling 125 Mb. The genome of an affected dog was sequenced and compared to 795 control genomes. The prioritization of private variants revealed a clear top candidate variant for the observed dwarfism. This variant, PRKG2:XM_022413533.1:c.1634+1G>T, affects the splice donor site and is therefore predicted to disrupt the function of the PKRG2 gene encoding protein, kinase cGMP-dependent type 2, a known regulator of chondrocyte differentiation. The genotypes of the PRKG2 variant were perfectly associated with the phenotype in the studied family of dogs. PRKG2 loss-of-function variants were previously reported to cause disproportionate dwarfism in humans, cattle, mice, and rats. Together with the comparative data from other species, our data strongly suggest PRKG2:c.1634+1G>T to be a candidate causative variant for the observed dwarfism phenotype in Dogo Argentino dogs.


Asunto(s)
Proteína Quinasa Dependiente de GMP Cíclico Tipo II/genética , Enfermedades de los Perros/genética , Enanismo/genética , Predisposición Genética a la Enfermedad , Animales , Bovinos , Enfermedades de los Perros/patología , Perros , Enanismo/patología , Enanismo/veterinaria , Ligamiento Genético/genética , Genotipo , Humanos , Ratones , Mutación/genética , Linaje , Fenotipo , Isoformas de Proteínas/genética , Ratas
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