Heterozygous loss of SRRM1 may be associated with neurodevelopmental phenotypes and anomalies in cell growth and neurite morphology
By trio exome sequencing and international collaboration, we identified (de novo) truncating variants in SRRM1, encoding a key component of spliceosomes and for mRNA processing, in three individuals with variable neurodevelopmental phenotypes. Knockdown of SRRM1 in SKNBE2 cells by CRISPR/Cas9 editing and subsequent differentiation into neuron like cells resulted in impaired cell proliferation, migration, and neurite outgrowth. Furthermore, pan-neuronal or motoneuronal knockdown of the orthologue Srrm1 in Drosophila lead to reduced viability or impaired gross neurological function, respectively. Taken together, our observations support SRRM1 as a candidate gene for Neurodevelopmental Disorders.
Altay et al., Eur J Hum Genet. 2025