Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000002628 | SCV001406668 | pathogenic | Hereditary spastic paraplegia 6 | 2023-12-13 | criteria provided, single submitter | clinical testing | This sequence change replaces threonine, which is neutral and polar, with arginine, which is basic and polar, at codon 45 of the NIPA1 protein (p.Thr45Arg). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individual(s) with clinical features of hereditary spastic paraplegia (PMID: 14508710). It has also been observed to segregate with disease in related individuals. ClinVar contains an entry for this variant (Variation ID: 2520). An algorithm developed to predict the effect of missense changes on protein structure and function (PolyPhen-2) suggests that this variant is likely to be disruptive. Experimental studies have shown that this missense change affects NIPA1 function (PMID: 17166836, 19091982, 20816793). For these reasons, this variant has been classified as Pathogenic. |
Gene |
RCV004719611 | SCV005325109 | pathogenic | not provided | 2023-10-13 | criteria provided, single submitter | clinical testing | Published functional studies demonstrate a damaging effect and show that this variant affects normal protein function and has profound effects on cellular distributions (Goytain et al., 2007; Botzolakis et al., 2011); Not observed at significant frequency in large population cohorts (gnomAD); In silico analysis supports that this missense variant has a deleterious effect on protein structure/function; This variant is associated with the following publications: (PMID: 18191948, 19091982, 35181192, 35464835, 19620182, 22302102, 32384786, 22552817, 20816793, 17166836, 14508710) |
OMIM | RCV000002628 | SCV000022786 | pathogenic | Hereditary spastic paraplegia 6 | 2003-10-01 | no assertion criteria provided | literature only |