Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000049320 | SCV002228672 | pathogenic | Congenital neutropenia-myelofibrosis-nephromegaly syndrome | 2023-09-19 | criteria provided, single submitter | clinical testing | This sequence change replaces threonine, which is neutral and polar, with asparagine, which is neutral and polar, at codon 224 of the VPS45 protein (p.Thr224Asn). This variant is not present in population databases (gnomAD no frequency). This missense change has been observed in individuals with severe congenital neutropenia (PMID: 23599270, 23738510). It has also been observed to segregate with disease in related individuals. ClinVar contains an entry for this variant (Variation ID: 55906). Advanced modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) performed at Invitae indicates that this missense variant is expected to disrupt VPS45 protein function. Experimental studies have shown that this missense change affects VPS45 function (PMID: 23599270). For these reasons, this variant has been classified as Pathogenic. |
Revvity Omics, |
RCV000049320 | SCV004238396 | pathogenic | Congenital neutropenia-myelofibrosis-nephromegaly syndrome | 2023-06-19 | criteria provided, single submitter | clinical testing | |
OMIM | RCV000049320 | SCV000081752 | pathogenic | Congenital neutropenia-myelofibrosis-nephromegaly syndrome | 2013-07-04 | no assertion criteria provided | literature only |