Axons get ahead: Insights into axon guidance and congenital cranial dysinnervation disorders

Developmental Neurobiology
John Chilton, Sarah Guthrie

Abstract

Cranial nerves innervate head muscles in a well-characterized and highly conserved pattern. Identification of genes responsible for human congenital disorders of these nerves, combined with the analysis of their role in axonal development in animal models, has advanced understanding of how neuromuscular connectivity is established. Here, we focus on the ocular motor system, as an instructive example of the success of this approach in unravelling the aetiology of human strabismus. The discovery that ocular motility disorders can arise from mutations in transcription factors, including HoxA1, HoxB1, MafB, Phox2A, and Sall4, has revealed gene regulatory networks that pattern the brainstem and/or govern the differentiation of cranial motor neurons. Mutations in genes involved in axon growth and guidance disrupt specific stages of the extension and pathfinding of ocular motor nerves, and have been implicated in human strabismus. These genes encompass varied classes of molecule, from receptor complexes to dynamic effectors to cytoskeletal components, including Robo3/Rig1, Alpha2-chimaerin, Kif21A, TUBB2, and TUBB3. A current challenge is to understand the protein regulatory networks that link the cell surface to the cytoskeleton and ...Continue Reading

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Citations

May 2, 2017·Human Molecular Genetics·Mary C Whitman, Elizabeth C Engle
May 5, 2017·Developmental Neurobiology·Sarah Guthrie, Alain Chédotal
Nov 15, 2017·Acta Neuropathologica Communications·Kenichi NagataTakaomi C Saido
Aug 4, 2018·Neuroradiology·Shaimaa Abdelsattar MohammadGhada M H Abdel-Salam
Aug 31, 2020·American Journal of Medical Genetics. Part a·Siddaramappa J PatilKatta Mohan Girisha
Dec 2, 2020·Journal of Medical Genetics·Xin Cynthia YeWyeth W Wasserman

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