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Mendeliome v1.4897 SRRM4 Chirag Patel Classified gene: SRRM4 as Green List (high evidence)
Mendeliome v1.4897 SRRM4 Chirag Patel Gene: srrm4 has been classified as Green List (High Evidence).
Mendeliome v1.4896 SRRM4 Chirag Patel Classified gene: SRRM4 as Green List (high evidence)
Mendeliome v1.4896 SRRM4 Chirag Patel Gene: srrm4 has been classified as Green List (High Evidence).
Mendeliome v1.4895 SRRM4 Chirag Patel Marked gene: SRRM4 as ready
Mendeliome v1.4895 SRRM4 Chirag Patel Gene: srrm4 has been classified as Red List (Low Evidence).
Mendeliome v1.4895 SRRM4 Chirag Patel changed review comment from: 3 unrelated individuals with developmental delay, absent speech, intellectual impairment, epilepsy, early‑onset dystonia and chorea (6mths-20mths). Trio WES/WGS identified a de novo canonical splice‑donor variant in SRRM4 in each individual (c.464+2T>C or c.464+2T>A). SRRM4 is an exclusively neural-expressed splicing-factor gene. RNA-sequencing in patient fibroblasts revealed 2 variant-specific mutant SRRM4-mRNA isoforms (1 x isoform lacking exon 5, and 1 x isoform containing a 69-nucleotide elongation of exon 5) suggestive of a mechanism other than loss-of-function. Additionally, they uncovered altered splicing patterns of known SRRM4 downstream mRNA-substrates in patient cells compared to SRRM4 expression-activated control fibroblasts.
Sources: Literature; to: 3 unrelated individuals with developmental delay, absent speech, intellectual impairment, epilepsy, early‑onset dystonia and chorea (6mths-20mths). Trio WES/WGS identified a rare de novo canonical splice‑donor variant in SRRM4 in each individual (c.464+2T>C or c.464+2T>A). SRRM4 is an exclusively neural-expressed splicing-factor gene. RNA-sequencing in patient fibroblasts revealed 2 variant-specific mutant SRRM4-mRNA isoforms (1 x isoform lacking exon 5, and 1 x isoform containing a 69-nucleotide elongation of exon 5) suggestive of a mechanism other than loss-of-function. Additionally, they uncovered altered splicing patterns of known SRRM4 downstream mRNA-substrates in patient cells compared to SRRM4 expression-activated control fibroblasts.
Sources: Literature
Mendeliome v1.4895 SRRM4 Chirag Patel gene: SRRM4 was added
gene: SRRM4 was added to Mendeliome. Sources: Literature
Mode of inheritance for gene: SRRM4 was set to MONOALLELIC, autosomal or pseudoautosomal, NOT imprinted
Publications for gene: SRRM4 were set to 41958152
Phenotypes for gene: SRRM4 were set to Neurodevelopmental disorder, MONDO:0700092, SRRM4-related
Mode of pathogenicity for gene: SRRM4 was set to Other
Review for gene: SRRM4 was set to GREEN
Added comment: 3 unrelated individuals with developmental delay, absent speech, intellectual impairment, epilepsy, early‑onset dystonia and chorea (6mths-20mths). Trio WES/WGS identified a de novo canonical splice‑donor variant in SRRM4 in each individual (c.464+2T>C or c.464+2T>A). SRRM4 is an exclusively neural-expressed splicing-factor gene. RNA-sequencing in patient fibroblasts revealed 2 variant-specific mutant SRRM4-mRNA isoforms (1 x isoform lacking exon 5, and 1 x isoform containing a 69-nucleotide elongation of exon 5) suggestive of a mechanism other than loss-of-function. Additionally, they uncovered altered splicing patterns of known SRRM4 downstream mRNA-substrates in patient cells compared to SRRM4 expression-activated control fibroblasts.
Sources: Literature