Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Paris Brain Institute, |
RCV001391393 | SCV001451210 | pathogenic | Hereditary spastic paraplegia 3A | criteria provided, single submitter | clinical testing | ||
Labcorp Genetics |
RCV001391393 | SCV002299386 | uncertain significance | Hereditary spastic paraplegia 3A | 2025-01-12 | criteria provided, single submitter | clinical testing | This sequence change replaces glutamine, which is neutral and polar, with lysine, which is basic and polar, at codon 251 of the ATL1 protein (p.Gln251Lys). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individual(s) with autosomal dominant hereditary spastic paraplegia (PMID: 15596607, 34983064). It has also been observed to segregate with disease in related individuals. This variant is also known as 919C>A. ClinVar contains an entry for this variant (Variation ID: 989016). Invitae Evidence Modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) has been performed for this missense variant. However, the output from this modeling did not meet the statistical confidence thresholds required to predict the impact of this variant on ATL1 protein function. This variant disrupts the p.Gln251 amino acid residue in ATL1. Other variant(s) that disrupt this residue have been observed in individuals with ATL1-related conditions (PMID: 31236401), which suggests that this may be a clinically significant amino acid residue. In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. |
Inherited Neuropathy Consortium Ii, |
RCV001391393 | SCV004011955 | uncertain significance | Hereditary spastic paraplegia 3A | 2016-01-06 | no assertion criteria provided | literature only |