Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Institute of Human Genetics, |
RCV000855419 | SCV000994921 | uncertain significance | Autosomal recessive nonsyndromic hearing loss 1A | 2019-10-02 | criteria provided, single submitter | clinical testing | The c.586G>A GJB3-variant (p.Ala196Thr) is found at a relatively low frequency (gnomAD & ExAC population frequency: 0.0025%) within the general population and has a pathogenic computational verdict due to 10 pathogenic predictions from DANN, DEOGEN2, FATHMM, FATHMM-MKL, FATHMM-XF, LRT, M-CAP, MutationAssessor, MutationTaster and SIFT vs. 3 benign predictions from PROVEAN, PrimateAI and SIFT4G. In our facility the variant was found in compound heterozygous state with a pathogenic GJB2-Variant (c.101T>C / p.Met34Thr) in an affected patient with non-syndromic congenital bilateral deafness. Segregation analysis revealed the same variant combination in the unaffected, healthy father. Thus, we consider this variant a variant of unknown significance (VUS), with possibly benign character. The possibility of this variant being pathogenic in homozygous state can not be excluded. |
Ambry Genetics | RCV004029276 | SCV004876993 | uncertain significance | Inborn genetic diseases | 2023-12-18 | criteria provided, single submitter | clinical testing | The c.586G>A (p.A196T) alteration is located in exon 2 (coding exon 1) of the GJB3 gene. This alteration results from a G to A substitution at nucleotide position 586, causing the alanine (A) at amino acid position 196 to be replaced by a threonine (T). Based on insufficient or conflicting evidence, the clinical significance of this alteration remains unclear. |