
The advent of techniques for the derivation of human neural crest cells (hNCCs) from human pluripotent stem cells (hPSCs), has not only enabled the understanding of NCC development and disease in a human-specific context but also opened new opportunities for therapeutic applications. Thus, NCCs have been widely studied in animal models to elucidate their role in a range of neurocristopathies involving the craniofacial skeleton and the peripheral nervous system. They form ectodermal derivatives, such as sensory and enteric neurons, Schwann cells, as well as mesenchymal derivatives ( Le Douarin and Dupin, 2003). Neural crest cells (NCCs) are transient, migratory stem cells that originate from the neural tube and migrate to different embryonic tissues to give rise to a wide variety of cell types ( Le Douarin et al., 2004). We will also discuss the potential applications of these human specific NCC platforms, including the use of iPSCs for disease modeling and the potential of NCCs for future regenerative applications. Here, we review the various in vitro strategies that have been used to derive NCCs from hPSCs and to specify NCCs into cranial, trunk, and vagal subpopulations and their derivatives.

The use of induced pluripotent stem cells (iPSCs) derived from patients afflicted with neurocristopathies has also enabled the study of defective human NCC development using these in vitro platforms. These in vitro differentiation platforms are a valuable tool to gain a better understanding of the molecular mechanisms involved in human neural crest development. In the recent years, there have been efforts to study NCC development and disease in human specific models, with protocols being established to derive NCCs from human pluripotent stem cells (hPSCs), and to further differentiate these NCCs to neural, mesenchymal and other lineages. NCC development has historically been studied in a variety of animal models, including xenopus, chick and mouse. Defects in the development, differentiation and migration of NCCs give rise to a class of syndromes and diseases that are known as neurocristopathies.

Neural crest cells (NCCs) are a multipotent and migratory cell population in the developing embryo that contribute to the formation of a wide range of tissues.
