Key result
Homozygous p.A31P cMyBP-C mutation is linked to ~21-fold greater fHCM risk in Maine Coon cats.
Why the study?
Larger studies are needed to establish the clinical significance of heterozygosity or homozygosity for the p.A31P cMyBP-C mutation in Maine Coon cats regarding echocardiographic indices and fHCM development.
Does the p.A31P cMyBP-C mutation increase the risk of hypertrophic cardiomyopathy in Maine Coon cats?
Cohort (n=332)
Single-blind
No
Does the p.A31P cMyBP-C mutation increase the risk of hypertrophic cardiomyopathy in Maine Coon cats?
Odds Ratio: 21.6 (95% CI 7.01–66.2)
In young Maine Coon cats, being homozygous for the p.A31P cMyBP-C mutation is strongly associated with hypertrophic cardiomyopathy, whereas heterozygosity has very low penetrance.
Homozygosity for p.A31P strongly associated with early fHCM in Maine Coons; challenges autosomal-dominant model from single pedigrees and leaves heterozygote penetrance open.
BACKGROUND: In Maine Coon (MC) cats the c.91G > C mutation in the gene MYBPC3, coding for cardiac myosin binding protein C (cMyBP-C), is associated with feline hypertrophic cardiomyopathy (fHCM). The mutation causes a substitution of an alanine for a proline at residue 31 (p.A31P) of cMyBP-C. The pattern of inheritance has been considered autosomal dominant based on a single pedigree. However, larger studies are needed to establish the significance of cats being heterozygous or homozygous for the mutation with respect to echocardiographic indices and the probability of developing fHCM. The objective of the present study was to establish the clinical significance of being homozygous or heterozygous for the p.A31P cMyBP-C mutation in young to middle-aged cats. METHODS: The cohort consisted of 332 MC cats, 282 cats < 4 years (85%). All cats were examined by 2-D and M-mode echocardiography. DNA was extracted from blood samples or buccal swabs and screened for the p.A31P cMyBP-C mutation in exon 3 of the gene, using polymerase chain reaction followed by DNA sequencing. RESULTS: The fHCM prevalence was 6.3% in the cohort. Eighteen cats were homozygous and 89 cats were heterozygous for the mutation. The odds ratio for having fHCM for homozygous cats was 21.6 (95% confidence interval 7.01-66.2) - when the group of equivocal cats was categorized as non-affected. Overall, 50% of the cats that were homozygous for the mutation had fHCM. p.A31P heterozygosity was not associated with a significant odds ratio for fHCM. In cats in the 4 to 6 years of age range a similar, non significant, odds ratio was seen in heterozygous cats. Only two cats over four years were homozygous and both were diagnosed with fHCM. CONCLUSION: As there is no significant odds ratio associated with being heterozygous for the pA31P cMyBP-C mutation at this age, the mutation must have a very low penetrance in this group. From our data it would appear that most MC cats that develop fHCM due to the p.A31P mutation prior to the age of approximately 6 years do so because they are homozygous for this mutation.
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Godiksen et al. (2011) conducted a cohort in Feline hypertrophic cardiomyopathy (n=332). Homozygous p.A31P cMyBP-C mutation vs. Wild type (no mutation) was evaluated on Feline hypertrophic cardiomyopathy (fHCM) (OR 21.6, 95% CI 7.01-66.2). Homozygosity for the p.A31P cMyBP-C mutation in Maine Coon cats was associated with a significantly increased risk of feline hypertrophic cardiomyopathy (OR 21.6), whereas heterozygosity showed very low penetrance.
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