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Elizabeth Engle’s research on congenital cranial dysinnervation disorders (CCDDs) has brought to light details of the genetics and fundamental mechanisms behind cranial motor neuron and axon development and function. Engle and her team have used family cohorts and model organisms to investigate gene expression and its epigenetic control in developing motor neurons, wiring of neural circuits, and response of neurons to growth signals in the context of these disorders. Their current work to understand cranial motor neuron specification and axon connectivity may inform research on human cognitive disorders such as schizophrenia, autism, dyslexia, and depression.