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.