Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000494398 | SCV000583227 | likely pathogenic | not provided | 2017-05-30 | criteria provided, single submitter | clinical testing | The c.1325_1326delTC variant in the SLC52A3 gene has been reported previously in the homozygous state and in the presence of another heterozygous SLC52A3 variant, in two related individuals with clinical features that include hypotonia, brisk reflexes in the lower limbs, bulbar palsy, and respiratory difficulties (Green et al., 2010). The c.1325_1326delTC variant causes a frameshift starting with codon Leucine 442, changes this amino acid to a Arginine residue, and creates a premature Stop codon at position 64 of the new reading frame, denoted p.Leu442ArgfsX64. This frameshift variant replaces the typical last 28 amino acid residues in the SLC52A3 protein with 63 different amino acid residues. This change is expected to alter the normal structure and function of the resultant protein. The c.1325_1326delTC variant is not observed in large population cohorts (Lek et al., 2016; 1000 Genomes Consortium et al., 2015; Exome Variant Server). We interpret c.1325_1326delTC as a likely pathogenic variant. |
OMIM | RCV000000162 | SCV000020305 | pathogenic | Brown-Vialetto-Van Laere syndrome 1 | 2010-03-12 | no assertion criteria provided | literature only | |
Gene |
RCV000000162 | SCV000246229 | pathogenic | Brown-Vialetto-Van Laere syndrome 1 | 2015-03-17 | no assertion criteria provided | literature only |