Trimebutine maleate [2-dimethylamino-2-phenylbutyl 3,4,5-trimethoxybenzoic acid] has been demonstrated to be active for relieving abdominal pain and it is widely used for patients with irritable bowel syndrome. Adverse drug reactions are mostly mild and well-tolerated. To our knowledge, only two cases of trimebutine induced hypersensitivity have been reported, and both were delayed type reactions. Here, we report the first case of trimebutine maleate-induced anaphylaxis.