Background Neuronal migration is considered a key process in human brain development. However, direct observation of migrating human cortical neurons in the fetal brain is accompanied by ethical concerns and is a major obstacle in investigating human cortical neuronal migration. New method We established a novel system that enables direct visualization of migrating cortical neurons generated from human induced pluripotent stem cells (hiPSCs). Results We observed the migration of cortical neurons generated from hiPSCs derived from a control and from a patient with lissencephaly. Methods Our system needs no viable brain tissue, which is usually used in slice culture. Migratory behavior of human cortical neuron can be observed more easily and more vividly by its fluorescence and glial scaffold than that by earlier methods. Conclusions Our in vitro experimental system provides a new platform for investigating development of the human central nervous system and brain malformation.
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