Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV003064236 | SCV003442799 | pathogenic | Alpha-1-antitrypsin deficiency | 2023-05-02 | criteria provided, single submitter | clinical testing | Variants that disrupt the consensus splice site are a relatively common cause of aberrant splicing (PMID: 17576681, 9536098). Algorithms developed to predict the effect of sequence changes on RNA splicing suggest that this variant may disrupt the consensus splice site. For these reasons, this variant has been classified as Pathogenic. Studies have shown that this variant does not significantly alter or has an unclear effect on SERPINA1 gene expression (PMID: 12220457). ClinVar contains an entry for this variant (Variation ID: 2137619). This variant has been observed in individual(s) with clinical features consistent with SERPINA1 related conditions (PMID: 12220457; Invitae). In at least one individual the data is consistent with being in trans (on the opposite chromosome) from a pathogenic variant. This variant is not present in population databases (gnomAD no frequency). This variant occurs in a non-coding region of the SERPINA1 gene. It does not change the encoded amino acid sequence of the SERPINA1 protein. It affects a nucleotide within the consensus splice site. |
Baylor Genetics | RCV003064236 | SCV004203119 | pathogenic | Alpha-1-antitrypsin deficiency | 2024-03-23 | criteria provided, single submitter | clinical testing | |
Laboratory for Molecular Medicine, |
RCV003064236 | SCV004847941 | likely pathogenic | Alpha-1-antitrypsin deficiency | 2015-04-03 | criteria provided, single submitter | clinical testing | The c.-5+1G>A variant in SERPINA1 has been previously reported in 1 compound heterozygous individual with emphysema and in 1 compound heterozygous individual with chronic obstructive pulmonary disease (COPD), and segregated with disease in 1 compound heterozygous individual with COPD (Seixas 2002, Lara 2014). Data from large population studies is insufficient to assess the frequency of this variant. This variant occurs in the invariant region (+/- 1, 2) of the splice consensus sequence of a non-coding exon and functional studies indicate this variant leads to altered splicing which is predicted to lead to an abnormal or absent protein (Lara 2014). In summary, although additional studies are required to fully establish its clinical significance, the c.-5+1G>A variant is likely pathogenic. |