Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000704798 | SCV000833765 | uncertain significance | Hypodontia | 2018-07-25 | criteria provided, single submitter | clinical testing | This sequence change replaces isoleucine with phenylalanine at codon 87 of the PAX9 protein (p.Ile87Phe). The isoleucine residue is highly conserved and there is a small physicochemical difference between isoleucine and phenylalanine. In summary, the available evidence is currently insufficient to determine the role of this variant in disease. Therefore, it has been classified as a Variant of Uncertain Significance. Experimental studies have shown that this missense change impairs protein stability and DNA binding activities of the PAX9 protien (PMID: 16479262). This variant has been observed to segregate with oligodontia in one family (PMID: 16479262). ClinVar contains an entry for this variant (Variation ID: 13775). This variant is not present in population databases (ExAC no frequency). |
OMIM | RCV000014785 | SCV000035040 | pathogenic | Tooth agenesis, selective, 3 | 2009-08-01 | no assertion criteria provided | literature only |