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Intellectual disability v8.127 ATXN7L3 Achchuthan Shanmugasundram Tag gene-checked tag was added to gene: ATXN7L3.
Intellectual disability v8.97 ATXN7L3 Sarah Leigh Tag Q3_24_promote_green was removed from gene: ATXN7L3.
Intellectual disability v8.97 ATXN7L3 Sarah Leigh commented on gene: ATXN7L3: The rating of this gene has been updated to green following NHS Genomic Medicine Service approval.
Intellectual disability v8.97 ATXN7L3 Sarah Leigh Source NHS GMS was added to ATXN7L3.
Source Expert Review Green was added to ATXN7L3.
Rating Changed from Amber List (moderate evidence) to Green List (high evidence)
Intellectual disability v8.36 ATXN7L3 Achchuthan Shanmugasundram Tag Q3_24_NHS_review was removed from gene: ATXN7L3.
Tag Q3_24_MOI was removed from gene: ATXN7L3.
Intellectual disability v7.3 ATXN7L3 Sarah Leigh Classified gene: ATXN7L3 as Amber List (moderate evidence)
Intellectual disability v7.3 ATXN7L3 Sarah Leigh Gene: atxn7l3 has been classified as Amber List (Moderate Evidence).
Intellectual disability v7.2 ATXN7L3 Sarah Leigh Tag Q3_24_promote_green tag was added to gene: ATXN7L3.
Tag Q3_24_NHS_review tag was added to gene: ATXN7L3.
Tag Q3_24_MOI tag was added to gene: ATXN7L3.
Intellectual disability v7.2 ATXN7L3 Sarah Leigh gene: ATXN7L3 was added
gene: ATXN7L3 was added to Intellectual disability. Sources: Literature
Mode of inheritance for gene: ATXN7L3 was set to MONOALLELIC, autosomal or pseudoautosomal, imprinted status unknown
Publications for gene: ATXN7L3 were set to 38753057; 33731875
Phenotypes for gene: ATXN7L3 were set to syndromic neurodevelopmental disorder
Review for gene: ATXN7L3 was set to GREEN
Added comment: ATXN7L3 variants are not associated with a phenotype in OMIM or Gen2Phen. PMID: 38753057 reports five monoallelic ATXN7L3 variants in nine unrelated cases. The variants were de novo, where this could be established (8/9 cases). Common features in the cases were: global developmental delay (8/9), dysmorphic features (7/9), hypotonia (7/9), strabismus (4/6), abnormal brain MRI (6/8). ATXN7L3 protein levels were reduced and deubiquitylation was impaired, resulting in increased levels of histone H2Bub1 in the fibroblasts of an affected individual carrying the recurrent variant: NM_001382309.1: c.340C>T; p.(Arg114Ter). This finding was consistent with the increased H2Bub1 levels in Atxn7l3-null mouse embryos, who have developmental delay and embryonic lethality (PMID: 33731875).
Sources: Literature