Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Laboratory for Molecular Medicine, |
RCV000004868 | SCV000966905 | pathogenic | Acroerythrokeratoderma | 2018-08-29 | criteria provided, single submitter | clinical testing | The p.Trp15Arg variant in SLURP1 has been reported 13 homozygous and 3 compound heterozygous individuals with clinical features of Mal de Meleda and segregated with disease in 9 affected releatives from at least 2 families (Eckl 2003, Nelle n 2013, Zhao 2014). This variant has been identified in 19/125816 European chrom osomes by the Genome Aggregation Database (gnomAD, http://gnomad.broadinstitute. org) and has been reported in ClinVar (Variation ID 4605). In vitro functional s tudies provide some evidence that the p.Trp15Arg variant may impact protein func tion (Favre 2007). In summary, this variant meets criteria to be classified as p athogenic for autosomal recessive Mal de Meleda based upon segregation studies a nd functional evidence. ACMG/AMP criteria applied: PM3_Strong, PP1_Strong, PS3_S upporting. |
OMIM | RCV000004868 | SCV000025044 | pathogenic | Acroerythrokeratoderma | 2003-01-01 | no assertion criteria provided | literature only |