Wisdom Panel Research Team, Wisdom Panel, Kinship, Helsinki, Finland.
Wisdom Panel Research Team, Wisdom Panel, Kinship, Portland, Oregon, United States of America.
PLoS Genet. 2023 Feb 27;19(2):e1010651. doi: 10.1371/journal.pgen.1010651. eCollection 2023 Feb.
Hundreds of genetic variants implicated in Mendelian disease have been characterized in dogs and commercial screening is being offered for most of them worldwide. There is typically limited information available regarding the broader population frequency of variants and uncertainty regarding their functional and clinical impact in ancestry backgrounds beyond the discovery breed. Genetic panel screening of disease-associated variants, commercially offered directly to the consumer or via a veterinary clinician, provides an opportunity to establish large-scale cohorts with phenotype data available to address open questions related to variant prevalence and relevance. We screened the largest canine cohort examined in a single study to date (1,054,293 representative dogs from our existing cohort of 3.5 million; a total of 811,628 mixed breed dogs and 242,665 purebreds from more than 150 countries) to examine the prevalence and distribution of a total of 250 genetic disease-associated variants in the general population. Electronic medical records from veterinary clinics were available for 43.5% of the genotyped dogs, enabling the clinical impact of variants to be investigated. We provide detailed frequencies for all tested variants across breeds and find that 57% of dogs carry at least one copy of a studied Mendelian disease-associated variant. Focusing on a subset of variants, we provide evidence of full penetrance for 10 variants, and plausible evidence for clinical significance of 22 variants, on diverse breed backgrounds. Specifically, we report that inherited hypocatalasia is a notable oral health condition, confirm that factor VII deficiency presents as subclinical bleeding propensity and verify two genetic causes of reduced leg length. We further assess genome-wide heterozygosity levels in over 100 breeds, and show that a reduction in genome-wide heterozygosity is associated with an increased Mendelian disease variant load. The accumulated knowledge represents a resource to guide discussions on genetic test relevance by breed.
数百种与孟德尔疾病相关的遗传变异已在犬类中得到描述,并且全球范围内大多数遗传变异都提供了商业筛查。通常,关于这些变异在发现品种以外的更广泛人群中的频率的信息有限,并且关于它们在遗传背景中的功能和临床影响的不确定性。通过向消费者直接或通过兽医临床医生提供与疾病相关的变异的遗传面板筛查,为建立具有表型数据的大规模队列提供了机会,这些数据可用于解决与变异流行率和相关性相关的开放性问题。我们对迄今为止在一项研究中检查的最大犬类队列进行了筛查(从我们现有的 350 万只犬的队列中抽取了 1,054,293 只代表性犬;共有来自 150 多个国家的 811,628 只混合品种犬和 242,665 只纯种犬),以检查普通人群中总共 250 种与遗传疾病相关的变异的流行率和分布。为 43.5%的基因分型犬提供了兽医诊所的电子病历,从而能够研究变异的临床影响。我们为所有经过测试的变异提供了跨品种的详细频率,发现 57%的犬携带至少一份研究的与孟德尔疾病相关的变异。我们重点关注一组变体,为 10 个变体提供了完全外显率的证据,并为 22 个变体的临床意义提供了合理的证据,这些变体存在于不同的品种背景下。具体而言,我们报告说遗传性低碳酸血症是一种显著的口腔健康状况,证实了因子 VII 缺乏症表现为亚临床出血倾向,并验证了两种导致腿部长度缩短的遗传原因。我们进一步评估了 100 多个品种的全基因组杂合度水平,并表明全基因组杂合度的降低与孟德尔疾病变异负荷的增加有关。这些积累的知识代表了一个资源,可用于指导按品种讨论基因检测相关性。