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Recent evidence from systematic studies of animal models and human patients suggests that the neuromuscular junction (NMJ) is an important early target in MND, demonstrating functional and structural abnormalities in advance of pathological changes occurring in the motor neuron cell body. The ability to study pathological changes occurring at the NMJ in humans is therefore likely to be important for furthering our understanding of disease pathogenesis, and also for designing and testing new therapeutics. However, there are many practical and technical reasons why it is not possible to visualise or record from NMJs in pre\u2010 and early\u2010symptomatic MND patients <jats:italic>in vivo<\/jats:italic>. Other approaches are therefore required. The development of stem cell technologies has opened up the possibility of creating human NMJs <jats:italic>in vitro<\/jats:italic>, using pluripotent cells generated from healthy individuals and patients with MND. This review covers historical attempts to develop mature and functional NMJs <jats:italic>in vitro<\/jats:italic>, using co\u2010cultures of muscle and nerve from animals, and discusses how recent developments in the generation and specification of human induced pluripotent stem cells provides an opportunity to build on these previous successes to recapitulate human neuromuscular connectivity <jats:italic>in vitro<\/jats:italic>.<\/jats:p>","DOI":"10.1111\/j.1469-7580.2011.01459.x","type":"journal-article","created":{"date-parts":[[2011,12,3]],"date-time":"2011-12-03T11:32:54Z","timestamp":1322911974000},"page":"122-130","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":35,"title":["Using induced pluripotent stem cells (iPSC) to model human neuromuscular connectivity: promise or reality?"],"prefix":"10.1111","volume":"220","author":[{"given":"Sophie 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