Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
IIFP, |
RCV000204551 | SCV000262539 | pathogenic | Mucopolysaccharidosis, MPS-II | 2011-12-07 | criteria provided, single submitter | research | |
Labcorp Genetics |
RCV000204551 | SCV002234986 | pathogenic | Mucopolysaccharidosis, MPS-II | 2021-10-07 | criteria provided, single submitter | clinical testing | This variant is not present in population databases (ExAC no frequency). For these reasons, this variant has been classified as Pathogenic. Studies have shown that this variant is associated with skipping of exon 5 but is expected to preserve the integrity of the reading frame (PMID: 27896113). Experimental studies have shown that disruption of this splice site affects IDS protein function (PMID: 31877959). Disruption of this splice site has been observed in individual(s) with clinical features of mucopolysaccharidosis type II (PMID: 27896113, 27246110, 31877959, 9875019). ClinVar contains an entry for this variant (Variation ID: 221221). This sequence change affects a donor splice site in intron 5 of the IDS gene. RNA analysis indicates that this variant induces altered splicing and likely results in a shortened protein product. |
Laboratory of Diagnosis and Therapy of Lysosomal Disorders, |
RCV000204551 | SCV005089199 | likely pathogenic | Mucopolysaccharidosis, MPS-II | 2024-06-07 | criteria provided, single submitter | literature only | Null variant (PVS1_Strong), Absent from controls (or at low frequency) in gnomAD database (PM2_Moderate), Patient’s phenotype or family history highly specific for the disease (PP4_Moderate) |