A case of benign recurrent intrahepatic cholestasis was reported. A man had first experienced jaundice at the age of twenty-five, and suffered three subsequent attacks over the next five years. These attacks were characterized by prodromal severe pruritus. During the icteric phase, serum levels of total bile acids increased, most being conjugates of primary bile acids. Electron microscopy revealed that the bile canaliculi were filled with an increased volume of granular substances. Some bile canaliculi fused together resulting in the formation of abnormally elongated channels which abutted on the Disse space. These observations suggest bile regurgitation through the channel of altered canaliculi. In convalescence, these bile canaliculi disappeared. Every attack was followed by complete recovery of liver structure and function.