Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Lupski Lab, |
RCV000119274 | SCV000256735 | pathogenic | Visceral myopathy 1 | 2014-03-27 | criteria provided, single submitter | research | |
New York Genome Center | RCV001535397 | SCV005044136 | likely pathogenic | Megacystis-microcolon-intestinal hypoperistalsis syndrome 5 | 2022-09-08 | criteria provided, single submitter | clinical testing | The inherited c.400T>A variant has previously been reported as de novo in an individual with prune belly syndrome, MMIHS, and megacystis [PMID:24676022]. The c.400T>A variant is absent in population databases (gnomAD v2.1.1 and v3.1.2, TOPMed Freeze 8, All of US), suggesting it is not a common benign variant in the populations represented in those databases. The c.400T>A variant is located in exon 5 of this 9-exon gene. In silico predictions are in favor of damaging effect for p.(Tyr134Asn) [(CADD v1.6 = 28.6, REVEL = 0.978)]; however, there are no functional studies to support or refute these predictions. The variant is reported in ClinVar [ClinVar ID:132805] as pathogenic in association with Visceral myopathy. Based on available evidence, this inherited c.400T>A, p.Tyr134Asn variant identified in ACTG2 is classified as Likely Pathogenic. |
OMIM | RCV001535397 | SCV000154709 | pathogenic | Megacystis-microcolon-intestinal hypoperistalsis syndrome 5 | 2014-03-01 | no assertion criteria provided | literature only |