Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Institute of Human Genetics, |
RCV000059576 | SCV001934370 | likely pathogenic | Lowe syndrome | 2020-12-11 | criteria provided, single submitter | clinical testing | This variant was identified as hemizygous. |
Women's Health and Genetics/Laboratory Corporation of America, |
RCV003488373 | SCV004241538 | uncertain significance | not specified | 2023-12-15 | criteria provided, single submitter | clinical testing | Variant summary: OCRL c.1009C>T (p.Arg337Cys) results in a non-conservative amino acid change located in the Inositol polyphosphate-related phosphatase (IPR000300) of the encoded protein sequence. Four of five in-silico tools predict a damaging effect of the variant on protein function. The variant allele was found at a frequency of 5.5e-06 in 183438 control chromosomes (gnomAD). The available data on variant occurrences in the general population are insufficient to allow any conclusion about variant significance. c.1009C>T has been reported in the literature in an individual affected with Lowe Disease (example: Hichri_2011). At least one publication reports experimental evidence evaluating an impact on protein function. The most pronounced variant effect results in 10%-<30% of normal activity (example: Hichri_2011). The following publication has been ascertained in the context of this evaluation (PMID: 21031565). One submitter has cited clinical-significance assessments for this variant to ClinVar after 2014 and has classified the variant as likely pathogenic. Based on the evidence outlined above, the variant was classified as VUS-possibly pathogenic. |
Uni |
RCV000059576 | SCV000091108 | not provided | Lowe syndrome | no assertion provided | not provided |