Total submissions: 1
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Pittsburgh Clinical Genomics Laboratory, |
RCV004785160 | SCV005397723 | likely pathogenic | Intellectual developmental disorder with dysmorphic facies and behavioral abnormalities | 2024-06-28 | criteria provided, single submitter | clinical testing | This sequence variant is deletion of 15 nucleotides and insertion of 1 nucleotide beginning at coding position 792 in the FBXO11 gene; this results in removing the entire exon 6 canonical splice donor site. This variant is predicted to generate a non-functional allele through either the expression of a truncated protein or a loss of F-box protein 11 expression due to nonsense-mediated decay. This novel de novo variant is absent from ClinVar, publications, and the gnomAD v4.1.0 population database (0/~1609000 alleles). Studies examining the functional consequence of this variant have not been performed, to our knowledge. Haploinsufficiency in FBXO11 is a known mechanism of disease (PMID: 29796876, 30057029). Based upon the evidence, we consider this variant to be likely pathogenic. ACMG Criteria: PM2, PS2, PVS1 |