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Mendeliome v2.334 FSD1L Zornitza Stark edited their review of gene: FSD1L: Added comment: In 6 individuals from 4 families, the findings were isolated to RP, hence second OMIM# added.; Changed phenotypes: Neurodevelopmental disorder with seizures, spastic tetraparesis, and vision impairment, MIM# 621643, Retinitis pigmentosa 109, MIM# 621656
Mendeliome v2.182 FSD1L Zornitza Stark Phenotypes for gene: FSD1L were changed from Neurodevelopmental disorder, MONDO:0700092, FSD1L-related to Neurodevelopmental disorder with seizures, spastic tetraparesis, and vision impairment, MIM# 621643
Mendeliome v2.181 FSD1L Zornitza Stark Publications for gene: FSD1L were set to
Mendeliome v2.180 FSD1L Zornitza Stark edited their review of gene: FSD1L: Added comment: Now published, 11 probands from 6 families reported. Key clinical features include severe intellectual disability, spastic tetraparesis, reduced vision, and epilepsy, severe hydrocephalus, corpus callosum agenesis/hypoplasia, mild ventricular dilation, optic nerve hypoplasia, and white matter reduction.; Changed publications: 41720098
Mendeliome v2.180 FSD1L Zornitza Stark reviewed gene: FSD1L: Rating: GREEN; Mode of pathogenicity: None; Publications: ; Phenotypes: Neurodevelopmental disorder with seizures, spastic tetraparesis, and vision impairment, MIM# 621643; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Mendeliome v2.0 FSD1L Gene migrated from ENSG00000106701 to ENSG00000106701 (gene set migration)
Mendeliome v1.1023 FSD1L Zornitza Stark Marked gene: FSD1L as ready
Mendeliome v1.1023 FSD1L Zornitza Stark Gene: fsd1l has been classified as Green List (High Evidence).
Mendeliome v1.1023 FSD1L Zornitza Stark Phenotypes for gene: FSD1L were changed from Neurodevelopmental disorder to Neurodevelopmental disorder, MONDO:0700092, FSD1L-related
Mendeliome v1.981 FSD1L chirag patel Classified gene: FSD1L as Green List (high evidence)
Mendeliome v1.981 FSD1L chirag patel Gene: fsd1l has been classified as Green List (High Evidence).
Mendeliome v1.980 FSD1L chirag patel gene: FSD1L was added
gene: FSD1L was added to Mendeliome. Sources: Other
Mode of inheritance for gene: FSD1L was set to BIALLELIC, autosomal or pseudoautosomal
Phenotypes for gene: FSD1L were set to Neurodevelopmental disorder
Review for gene: FSD1L was set to GREEN
gene: FSD1L was marked as current diagnostic
Added comment: ESHG 2023:
8 families with biallelic missense/nonsense variants
Presentation only described 1 family/2 affecteds with DD, ID, spastic paraparesis, epilepsy, corpus callosum hypoplasia, and optic nerve hypoplasia

Functional assays:
-reduced expression of FSD1L in mature neurons (RNA studies)
-very low % mature neurons (neuronal differentiation)
-reduced neuronal migration
Sources: Other