Litcius/Paper detail

Duplication/triplication mosaicism of EBF3 and expansion of the EBF3 neurodevelopmental disorder phenotype

Erika Ignatius, Riina Puosi, Maarit Palomäki, Noora Forsbom, Max Pohjanpelto, Tiina Alitalo, Anna‐Kaisa Anttonen, Kristiina Avela, Leena Haataja, Christopher J. Carroll, Tuula Lönnqvist, Pirjo Isohanni

2021European Journal of Paediatric Neurology14 citationsDOIOpen Access PDF

Abstract

Deleterious variants in the transcription factor early B-cell factor 3 (EBF3) are known to cause a neurodevelopmental disorder (EBF3-NDD). We report eleven individuals with EBF3 variants, including an individual with a duplication/triplication mosaicism of a region encompassing EBF3 and a phenotype consistent with EBF3-NDD, which may reflect the importance of EBF3 gene-dosage for neurodevelopment. The phenotype of individuals in this cohort was quite mild compared to the core phenotype of previously described individuals. Although ataxia tended to wane with age, we show that cognitive difficulties may increase, and we recommend that individuals with EBF3-NDD have systematic neuropsychological follow-up.

Topics & Concepts

Neurodevelopmental disorderPhenotypeGene duplicationGeneticsBiologyGeneGenetics and Neurodevelopmental DisordersGenomic variations and chromosomal abnormalitiesGenomics and Rare Diseases