Zic2 is required for enteric nervous system development and neurite outgrowth: a mouse model of enteric hyperplasia and dysplasia.

From a forward genetic screen in mice we isolated a novel Zic2 allele with abnormal aggregation of meconium in the gastrointestinal tract. Zic2(m1Nisw) mutant embryos show an increase in the number of enteric neurons in vivo and disorganization of the neurite network. Explant culture of Zic2(m1Nisw) gastrointestinal tract show extensive neurite outgrowth, suggesting that Zic2 is a negative regulator of nerve fiber growth. These studies demonstrate a previously unknown function of Zic2 and provide a novel animal model of enteric nervous system dysplasia and hyperplasia.