Herein, 2 cases of megacystis secondary to myenteric plexus pathology are reported. Unlike the entity of visceral myopathy, there was ganglion cell loss both in the bladder and in the colon of these 2 cases. Moreover, they did not present any gastrointestinal symptoms. Megacystis was the common pathology in these patients, whereas 1 of them suffered from unilateral vesicoureteral reflux. We could not find any similar report dealing with this phenomenon up to this time in the literature. Copyright (C) 2001 S. Karger AG, Basel.