Total submissions: 4
Submitter | RCV | SCV | Clinical significance | Condition | Last evaluated | Review status | Method | Comment |
---|---|---|---|---|---|---|---|---|
SIB Swiss Institute of Bioinformatics | RCV000853217 | SCV001245386 | uncertain significance | Neurodevelopmental disorder with microcephaly, arthrogryposis, and structural brain anomalies | 2020-02-14 | criteria provided, single submitter | curation | This variant is interpreted as a variant of uncertain significance for Neurodevelopmental disorder with microcephaly, arthrogryposis, and structural brain anomalies, autosomal recessive. The following ACMG Tag(s) were applied: PM2, PP3, PP1. |
Broad Center for Mendelian Genomics, |
RCV000853217 | SCV001430802 | uncertain significance | Neurodevelopmental disorder with microcephaly, arthrogryposis, and structural brain anomalies | 2020-05-29 | criteria provided, single submitter | research | The homozygous p.Pro446Leu variant in SMPD4 was identified by our study in 1 individual with neurodevelopmental disorder with microcephaly, arthrogryposis, and structural brain anomalies (PMID: 31495489). The variant has been identified in 0.006% (2/35360) of Latino chromosomes by the Genome Aggregation Database (gnomAD, http://gnomad.broadinstitute.org). Although this variant has been seen in the general population, its frequency is low enough to be consistent with a recessive carrier frequency. This variant has also been reported in ClinVar (Variation ID#: 691972) as pathogenic by OMIM. Computational prediction tools and conservation analyses do not provide strong support for or against an impact to the protein. In summary, while there is some suspicion for a pathogenic role, the clinical significance of this variant is uncertain. ACMG/AMP Criteria applied: PM2, PM3_supporting (Richards 2015). |
Genomic Medicine Center of Excellence, |
RCV000853217 | SCV004808122 | uncertain significance | Neurodevelopmental disorder with microcephaly, arthrogryposis, and structural brain anomalies | 2024-03-29 | criteria provided, single submitter | clinical testing | |
OMIM | RCV000853217 | SCV000996029 | pathogenic | Neurodevelopmental disorder with microcephaly, arthrogryposis, and structural brain anomalies | 2019-10-16 | no assertion criteria provided | literature only |