Total submissions: 8
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
Labcorp Genetics |
RCV001370842 | SCV001567385 | pathogenic | not provided | 2025-01-29 | criteria provided, single submitter | clinical testing | This sequence change replaces valine, which is neutral and non-polar, with glutamic acid, which is acidic and polar, at codon 408 of the SLC34A1 protein (p.Val408Glu). This variant is present in population databases (rs140649226, gnomAD 0.02%). This missense change has been observed in individual(s) with infantile hypercalcemia and/or kidney disease (PMID: 26047794, 31672324; internal data). In at least one individual the data is consistent with being in trans (on the opposite chromosome) from a pathogenic variant. ClinVar contains an entry for this variant (Variation ID: 1061290). Invitae Evidence Modeling of protein sequence and biophysical properties (such as structural, functional, and spatial information, amino acid conservation, physicochemical variation, residue mobility, and thermodynamic stability) indicates that this missense variant is not expected to disrupt SLC34A1 protein function with a negative predictive value of 80%. Experimental studies have shown that this missense change affects SLC34A1 function (PMID: 26047794). For these reasons, this variant has been classified as Pathogenic. |
Gene |
RCV001370842 | SCV001777647 | uncertain significance | not provided | 2021-04-23 | criteria provided, single submitter | clinical testing | Published functional studies suggest V408E causes a loss of phosphate transport activity (Schlingmann et al., 2016); In silico analysis, which includes protein predictors and evolutionary conservation, supports a deleterious effect; This variant is associated with the following publications: (PMID: 31188746, 31672324, 29275531, 26047794) |
Ce |
RCV001370842 | SCV003917016 | likely pathogenic | not provided | 2023-09-01 | criteria provided, single submitter | clinical testing | SLC34A1: PM3:Strong, PM2, PS3:Supporting |
ARUP Laboratories, |
RCV001370842 | SCV004564029 | uncertain significance | not provided | 2023-06-28 | criteria provided, single submitter | clinical testing | |
Institute of Human Genetics, |
RCV003983880 | SCV005368075 | likely pathogenic | Hypercalcemia, infantile, 2 | 2024-04-08 | criteria provided, single submitter | clinical testing | Criteria applied: PS3_MOD,PM3,PM5_SUP,PP3 |
Fulgent Genetics, |
RCV005040226 | SCV005671556 | likely pathogenic | Hypophosphatemic nephrolithiasis/osteoporosis 1; Fanconi renotubular syndrome 2; Hypercalcemia, infantile, 2 | 2024-04-17 | criteria provided, single submitter | clinical testing | |
Molecular Genetics Laboratory, |
RCV003983880 | SCV004708194 | pathogenic | Hypercalcemia, infantile, 2 | 2024-03-08 | no assertion criteria provided | clinical testing | |
Prevention |
RCV004734151 | SCV005361241 | pathogenic | SLC34A1-related disorder | 2024-05-30 | no assertion criteria provided | clinical testing | The SLC34A1 c.1223T>A variant is predicted to result in the amino acid substitution p.Val408Glu. This variant was reported in the compound heterozygous state in an individual with idiopathic infantile hypercalcemia (Schlingmann et al. 2016. PubMed ID: 26047794). This variant was also reported in the heterozygous state in an individual with renal hypophosphatemia (Table S1, Hureaux et al. 2019. PubMed ID: 31672324). Functional studies showed that this variant causes reduced SLC34A1 trafficking to the plasma membrane and loss of phosphate transport activity (Schlingmann et al. 2016. PubMed ID: 26047794). This variant is reported in 0.019% of alleles in individuals of European (Non-Finnish) descent in gnomAD. A different nucleotide substitution affecting the same amino acid (p.Val408Met) is associated with renal hypophosphatemia (Table S1, Hureaux et al. 2019. PubMed ID: 31672324). Taken together, the c.1223T>A (p.Val408Glu) is interpreted as pathogenic. |