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Reported is a rare superficial papillary adenocarcinoma arising from a congenital cyst, along with a survey of pertinent cases in the world literature. A 43-year-old male with a huge hepatic cyst was admitted to hospital for further examination. Since the cystic drainage done for alcoholic fixation was seromucinous initially but later contaminated by bile, an operation was initiated for dome resection and the closure of the biliary communication. At the time of the deroofing, however, small granulomas were found. And those were confirmed as being malignant by frozen section. Thus, an extended right lobectomy was performed for therapy. From its history and nature, this was considered to be a cancer arising in a congenital hepatic cyst.
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