Total submissions: 3
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV000632720 | SCV000753906 | pathogenic | Neuronal ceroid lipofuscinosis | 2023-11-19 | criteria provided, single submitter | clinical testing | This variant, c.346_348del, results in the deletion of 1 amino acid(s) of the DNAJC5 protein (p.Leu116del), but otherwise preserves the integrity of the reading frame. This variant is not present in population databases (gnomAD no frequency). This variant has been observed in individual(s) with adult-onset neuronal ceroid lipofuscinosis (PMID: 21820099, 22073189, 22235333, 22978711). It has also been observed to segregate with disease in related individuals. ClinVar contains an entry for this variant (Variation ID: 30893). Algorithms developed to predict the effect of variants on protein structure and function are not available or were not evaluated for this variant. Experimental studies have shown that this variant affects DNAJC5 function (PMID: 21820099, 22902780). For these reasons, this variant has been classified as Pathogenic. |
Research Unit for Rare Diseases, |
RCV000023878 | SCV000999259 | pathogenic | Ceroid lipofuscinosis, neuronal, 4 (Kufs type) | 2019-09-01 | criteria provided, single submitter | research | The c.343_345del (p.(L116del)), in exon 4 of DNAJC5 (NM_025219.2) variant was reported by Noskova et al. in 2011 as causative for autosomal dominant adult-onset neuronal ceroid lipofuscinosis (DOI: 10.1016/j.ajhg.2011.07.003). |
OMIM | RCV000023878 | SCV000045169 | pathogenic | Ceroid lipofuscinosis, neuronal, 4 (Kufs type) | 2013-06-01 | no assertion criteria provided | literature only |