Total submissions: 2
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Gene |
RCV000255314 | SCV000321783 | pathogenic | not provided | 2015-08-28 | criteria provided, single submitter | clinical testing | The C534X variant has been reported previously in association with leukocyte adhesion deficiency (LAD-1) (López-RodrÃguez et al., 1993; Roos et al., 1993). This variant is also predicted to cause loss of normal protein function either through protein truncation or nonsense-mediated mRNA decay. In addition, studies of the C534X variant suggest that it decreases mRNA expression (Shaw et al., 2001). |
Labcorp Genetics |
RCV003522952 | SCV004353298 | pathogenic | Leukocyte adhesion deficiency 1 | 2023-08-21 | criteria provided, single submitter | clinical testing | For these reasons, this variant has been classified as Pathogenic. ClinVar contains an entry for this variant (Variation ID: 265200). This sequence change creates a premature translational stop signal (p.Cys534*) in the ITGB2 gene. It is expected to result in an absent or disrupted protein product. Loss-of-function variants in ITGB2 are known to be pathogenic (PMID: 22134107, 25703682). This variant is present in population databases (no rsID available, gnomAD 0.009%). This premature translational stop signal has been observed in individual(s) with leukocyte adhesion deficiency (PMID: 7901025, 11882363). |