Test Detail

GM2 gangliosidosis (Sandhoff / Tay-Sachs)

Metabólico · Dog

Lysosomal storage disorder due to beta-hexosaminidase deficiency that causes accumulation of GM2 gangliosides in the nervous system. The most frequent form in the dog is variant 0 (Sandhoff), due to variants in HEXB that affect hexosaminidase A and B; in the Japanese Chin a Tay-Sachs-like form (type I / variant B) due to variants in HEXA has been described. It produces progressive neurodegeneration of juvenile onset with a lethal course. It is inherited in an autosomal recessive manner.
Inheritance patternAutosomal recessive (OMIA:001462-9615 and OMIA:001461-9615).
Gene / MutationSandhoff (variant 0), HEXB gene (CFA2): Toy Poodle, c.391del (p.Val131*) —published as c.283delG p.(Val59fs)— (OMIA:001462-9615; PMID 22766310). Shiba Inu, in-frame deletion c.849_851del (p.Leu284del) —published as c.618-620delCCT p.Leu207del— (PMID 28833537 and 29106755). Tay-Sachs (type I / variant B), HEXA gene (CFA30): Japanese Chin, c.967G>A, p.(Glu323Lys) (OMIA:001461-9615; PMID 23266199).
PenetranceLethal penetrance in homozygotes; heterozygotes are asymptomatic carriers.
Codejuiq
Turnaround time15 days
Price52,60 €

Incidence

Breeds with a documented variant: Toy Poodle (HEXB; the allele is rare in the population, Rahman 2012), Shiba Inu (HEXB; rare but distributed variant, Kolicheski 2017 and Wang 2018) and Japanese Chin (HEXA c.967G>A; identified in two related dogs, Sanders 2013). Limited data on reliable frequencies outside these series.

Breeder management

- Test breeding animals with the breed-specific test (HEXB in Toy Poodle and Shiba Inu; HEXA in Japanese Chin depending on the laboratory)
- Do not mate two carriers: 25% risk of lethal affected homozygotes
- A carrier can be mated with a free animal; offspring intended for breeding must be tested
- In the Shiba Inu, also consider the GM1 test (GLB1) because both diseases coexist
- Exclude affected animals from breeding

Specialist notes

Differential diagnosis with GM1 (GLB1) of the Shiba, almost clinically indistinguishable, and with other neurodegenerations of the young. The hexosaminidase A and B activity assay in plasma/leukocytes gives an indication of the variant (Sandhoff vs Tay-Sachs), but confirmation is molecular. MRI shows cerebral and cerebellar atrophy with white matter involvement.

References

1. Rahman MM et al. (2012) A frameshift mutation in the canine HEXB gene in toy poodles with GM2 gangliosidosis variant 0 (Sandhoff disease). Vet J 194(3):412-416. PMID: 22766310
2. Kolicheski AL et al. (2017) GM2 gangliosidosis in Shiba Inu dogs with an in-frame deletion in HEXB. J Vet Intern Med 31(5):1520-1526. PMID: 28833537
3. Wang P et al. (2018) Canine GM2-gangliosidosis Sandhoff disease associated with a 3-base pair deletion in the HEXB gene. J Vet Intern Med 32(1):340-347. PMID: 29106755
4. Sanders DN et al. (2013) GM2 gangliosidosis associated with a HEXA missense mutation in Japanese Chin dogs: a potential model for Tay Sachs disease. Mol Genet Metab 108(1):70-75. PMID: 23266199

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