Total submissions: 6
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Baylor Genetics | RCV000119282 | SCV001519924 | pathogenic | Pontocerebellar hypoplasia type 9 | 2022-02-21 | criteria provided, single submitter | clinical testing | |
Labcorp Genetics |
RCV002515805 | SCV002979315 | likely pathogenic | Hereditary spastic paraplegia 63; Pontocerebellar hypoplasia type 9 | 2023-06-29 | criteria provided, single submitter | clinical testing | In summary, the currently available evidence indicates that the variant is pathogenic, but additional data are needed to prove that conclusively. Therefore, this variant has been classified as Likely Pathogenic. Experimental studies have shown that this missense change affects AMPD2 function (PMID: 23911318). This sequence change replaces arginine, which is basic and polar, with histidine, which is basic and polar, at codon 674 of the AMPD2 protein (p.Arg674His). This variant is present in population databases (rs587777395, gnomAD 0.005%). This missense change has been observed in individuals with pontocerebellar hypoplasia (PMID: 23911318, 31130284). It has also been observed to segregate with disease in related individuals. This variant is also known as NM_203404.1:c.1664G>A (p.Arg555His). ClinVar contains an entry for this variant (Variation ID: 132813). 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. |
Genome- |
RCV003343648 | SCV004050605 | likely pathogenic | Hereditary spastic paraplegia 63 | 2023-04-11 | criteria provided, single submitter | clinical testing | |
Genome- |
RCV000119282 | SCV004050606 | likely pathogenic | Pontocerebellar hypoplasia type 9 | 2023-04-11 | criteria provided, single submitter | clinical testing | |
Genomic Medicine Center of Excellence, |
RCV000119282 | SCV004801197 | pathogenic | Pontocerebellar hypoplasia type 9 | 2024-03-14 | criteria provided, single submitter | research | |
OMIM | RCV000119282 | SCV000154717 | pathogenic | Pontocerebellar hypoplasia type 9 | 2013-08-01 | no assertion criteria provided | literature only |