Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Department of Genetics, |
RCV000191977 | SCV000891684 | pathogenic | Brown-Vialetto-Van Laere syndrome 1 | 2017-12-30 | criteria provided, single submitter | curation | |
Laboratory for Molecular Medicine, |
RCV001195405 | SCV001365755 | uncertain significance | not specified | 2019-08-27 | criteria provided, single submitter | clinical testing | The p.Arg212Cys with SLC52A3 has been previously reported in 3 homozygous individuals of Arab decent with suspected Brown-Vialetto-Van-Laere syndrome; however, it was not clear if these individuals were related (Manole 2017). In the same study, the variant was also reported in the homozygous state in one individual who was unaffected at the age of 12 years. The variant has also been identified in 0.002% (2/111872) of European chromosomes by gnomAD (http://gnomad.broadinstitute.org). Computational prediction tools and conservation analyses suggest that this variant may not impact the protein. Of note, three mammals (opossum, Tasmanian devil, platypus) harbor a cysteine (Cys) at this position, suggesting that this change may be tolerated. In summary, the clinical significance of this variant is uncertain due to the presence of conflicting data. ACMG/AMP criteria applied: PM2, PM3_Supporting, BP4. |
Gene |
RCV000191977 | SCV000246233 | pathogenic | Brown-Vialetto-Van Laere syndrome 1 | 2015-03-17 | no assertion criteria provided | literature only |