Total submissions: 5
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Ambry Genetics | RCV000623340 | SCV000742819 | pathogenic | Inborn genetic diseases | 2017-08-02 | criteria provided, single submitter | clinical testing | |
Illumina Laboratory Services, |
RCV000789766 | SCV004035998 | uncertain significance | Giant axonal neuropathy 1 | 2023-01-26 | criteria provided, single submitter | clinical testing | The GAN c.806G>A (p.Arg269Gln) missense variant results in the substitution of arginine at amino acid position 269 with glutamine. This variant has been reported in a homozygous state in two siblings with giant axonal neuropathy from reportedly non-consanguineous parents from the same village (PMID: 12655563). A different amino acid change at the same position, c.805C>T (p.Arg269Trp), was also reported in a compound heterozygous state with a missense variant of unknown significance in an affected female with an atypical giant axonal neuropathy phenotype (PMID: 23248352). The c.806G>A variant is reported in the Genome Aggregation Database in three alleles at a frequency of 0.000011 in the total population (version 2.1.1). Based on the available evidence, the c.806G>A (p.Arg269Gln) variant is classified as a variant of uncertain significance for giant axonal neuropathy. |
Concord Molecular Medicine Laboratory, |
RCV000789766 | SCV004562016 | likely pathogenic | Giant axonal neuropathy 1 | 2024-02-14 | criteria provided, single submitter | clinical testing | This variant is detected in homozyous state in a patient with a clinical diagnosis of giant axonal neuropathy, with the typical associated fizzy hair. This variant is present at extremely low frequency in population database (gnomAD v4 0.0005%). It has been reported in two siblings in a homozygous state with giant axonal neuropathy (PMID: 12655563). In silico analysis predicts this variant to be damaging (REVEL 0.78). A different amino acid change at the same position, p.(Arg269Trp), was also reported in a compound heterozygous state in patients with GAN-related conditions (PMID: 23248352, 23890932, 32999401). |
Inherited Neuropathy Consortium | RCV000789766 | SCV000929147 | likely benign | Giant axonal neuropathy 1 | flagged submission | literature only | ||
Inherited Neuropathy Consortium Ii, |
RCV000789766 | SCV004174498 | uncertain significance | Giant axonal neuropathy 1 | 2016-01-06 | no assertion criteria provided | literature only |