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Microcephaly v1.424 RNU6ATAC Zornitza Stark Phenotypes for gene: RNU6ATAC were changed from Neurodevelopmental disorder (MONDO:0700092), RNU6ATAC-related; neonatal diabetes to Syndromic disease, MONDO:0002254, RNU6ATAC-related
Microcephaly v1.423 RNU6ATAC Zornitza Stark Publications for gene: RNU6ATAC were set to 40975062
Microcephaly v1.422 RNU6ATAC Zornitza Stark Classified gene: RNU6ATAC as Green List (high evidence)
Microcephaly v1.422 RNU6ATAC Zornitza Stark Gene: rnu6atac has been classified as Green List (High Evidence).
Microcephaly v1.421 RNU6ATAC Zornitza Stark edited their review of gene: RNU6ATAC: Added comment: PMID 40975062 reports 1 individual from with biallelic RNU6ATAC variants causing childhood‑onset growth restriction, microcephaly, epilepsy, intellectual disability and ataxia. PMID 41808409 reports 3 unrelated families (3 individuals) with biallelic loss‑of‑function RNU6ATAC variants presenting with short stature, neurodevelopmental delay, epilepsy, immunodeficiency, skeletal dysplasia and other multisystem features; RNA‑seq shows marked excess of minor intron retention, confirming spliceosome dysfunction.

However, note overlap in authors between the two papers ?double-counting.

PMID 41864208 -- published version of the preprint referenced in previous reviews. 7 individuals from 4 families with early-onset diabetes (diagnosed aged <5 years) and immune dysregulatory features caused by bi-allelic variants in RNU6ATAC.

Genotype-phenotype correlation unclear at this stage but note that of the three individuals reported in 41808409, one did not have ID/neurological features but all had immunological involvement.; Changed rating: GREEN; Changed publications: 41808409, 40975062, 41864208; Changed phenotypes: Syndromic disease, MONDO:0002254, RNU6ATAC-related
Microcephaly v1.408 RNU6ATAC Chirag Patel Phenotypes for gene: RNU6ATAC were changed from Neurodevelopmental disorder (MONDO:0700092), RNU6ATAC-related to Neurodevelopmental disorder (MONDO:0700092), RNU6ATAC-related; neonatal diabetes
Microcephaly v1.407 RNU6ATAC Chirag Patel Tag non-coding gene tag was added to gene: RNU6ATAC.
Microcephaly v1.407 Chirag Patel Added reviews for gene RNU6ATAC from panel Mendeliome
Microcephaly v1.406 RNU6ATAC Chirag Patel Classified gene: RNU6ATAC as Amber List (moderate evidence)
Microcephaly v1.406 RNU6ATAC Chirag Patel Gene: rnu6atac has been classified as Amber List (Moderate Evidence).
Microcephaly v1.346 RNU6ATAC Zornitza Stark Marked gene: RNU6ATAC as ready
Microcephaly v1.346 RNU6ATAC Zornitza Stark Gene: rnu6atac has been classified as Red List (Low Evidence).
Microcephaly v1.346 RNU6ATAC Zornitza Stark Classified gene: RNU6ATAC as Red List (low evidence)
Microcephaly v1.346 RNU6ATAC Zornitza Stark Gene: rnu6atac has been classified as Red List (Low Evidence).
Microcephaly v1.345 RNU6ATAC Zornitza Stark reviewed gene: RNU6ATAC: Rating: RED; Mode of pathogenicity: None; Publications: ; Phenotypes: ; Mode of inheritance: BIALLELIC, autosomal or pseudoautosomal
Microcephaly v1.345 RNU6ATAC Lucy Spencer gene: RNU6ATAC was added
gene: RNU6ATAC was added to Microcephaly. Sources: Literature
Mode of inheritance for gene: RNU6ATAC was set to BIALLELIC, autosomal or pseudoautosomal
Publications for gene: RNU6ATAC were set to 40975062
Phenotypes for gene: RNU6ATAC were set to Neurodevelopmental disorder (MONDO:0700092), RNU6ATAC-related
Review for gene: RNU6ATAC was set to AMBER
Added comment: PMID: 40975062 1 patient compound heterozygous for n.36T>G and n.28C>T. Has short stature, microcephaly, hypotonia, neurodevelopmental delay, ID, seizures, ataxia, ventriculomegaly, syndactyly, nystagmus and oculomotor apraxia. Identified in a cohort of individuals with an excess of significant intron retention outliers in minor intron containing genes which are usually removed by the minor spliceosome of which RNU6ATAC is a part (as is RNU4ATAC). Proband had no candidate variants in RNU4ATAC or RNU12. Both RNU6ATAC variants are in a highly conserved 39bp region, and affect nucleotides predicted to be important for binding to U4ATAC.
Sources: Literature