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BRIP1 has been evaluated as a candidate moderate-penetrance gene for autosomal dominant hereditary breast carcinoma. Two rare heterozygous variants have been reported in index patients: a truncating frameshift c.2992_2995del (p.Lys998fsTer) causing BRCA1-binding disruption and protein instability with loss of the wild-type allele in tumor tissue (PMID:18628483), and a missense c.550G>T (p.Asp184Tyr) that creates a novel splice enhancer leading to exon 5 skipping and predicted helicase domain loss (PMID:30230034). No extended family segregation has been documented.
However, large-scale analyses have not supported a substantial role for BRIP1 in breast cancer predisposition. In 111 Finnish families, multiplex ligation-dependent probe amplification found no exonic rearrangements in BRIP1 (PMID:20567916), and German case-control studies of Pro919Ser and –64G>A variants showed no significant risk differences (PMID:17504528).
Gene–Disease AssociationLimitedSingle-family reports of two pathogenic BRIP1 variants with no segregation ([PMID:30230034], [PMID:18628483]) contrasted by negative large familial and case-control studies ([PMID:20567916], [PMID:17504528]). Genetic EvidenceLimitedTwo probands with rare BRIP1 variants, no extended segregation data, total of two variant classes observed. Functional EvidenceModerateSplicing assay demonstrates exon 5 skipping for c.550G>T ([PMID:30230034]); truncation c.2992_2995del disrupts BRCA1 binding and protein stability in cell models ([PMID:18628483]). |