Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Women's Health and Genetics/Laboratory Corporation of America, |
RCV000781473 | SCV000919531 | pathogenic | Mucopolysaccharidosis, MPS-II | 2018-12-26 | criteria provided, single submitter | clinical testing | Variant summary: IDS c.1044C>G (p.Tyr348X) results in a premature termination codon, predicted to cause a truncation of the encoded protein or absence of the protein due to nonsense mediated decay, which are commonly known mechanisms for disease. The variant was absent in 87394 control chromosomes (ExAC). c.1044C>G has been reported in the literature in individuals affected with Mucopolysaccharidosis Type II (Hunter Syndrome) (Froissart_2007, Charoenwattanasatien_2012). These data indicate that the variant may be associated with disease. A functional study, showed the variant to have no IDS activity in COS7 cells (Charoenwattanasatien_2012). No clinical diagnostic laboratories have submitted clinical-significance assessments for this variant to ClinVar after 2014. Based on the evidence outlined above, the variant was classified as pathogenic. |
Revvity Omics, |
RCV000781473 | SCV002018180 | pathogenic | Mucopolysaccharidosis, MPS-II | 2021-07-24 | criteria provided, single submitter | clinical testing | |
Laboratory of Diagnosis and Therapy of Lysosomal Disorders, |
RCV000781473 | SCV005089415 | pathogenic | Mucopolysaccharidosis, MPS-II | 2024-06-07 | criteria provided, single submitter | literature only | Null variant (PVS1_VeryStrong), In vitro or in vivo functional studies supportive of a damaging effect (PS3_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_Strong) |