Congenital liver cysts are not as rare as it was thought in the past. Thanks to new imaging diagnostic techniques, these cysts have been discovered with increasing frequency. Uncomplicated congenital liver cysts are usually asymptomatic or followed by mild symptoms. Various complications of these cysts were reported. Infection of the congenital liver cyst is, however, very rare complication. A 65-year old man with huge infected congenital central liver cyst containing 3400 ml of pus from which Escherichia coli was cultivated was presented. The patient had a number of other histologically confirmed congenital cysts of the liver and the cyst of the left kidney. The patient was cured by pus evacuation, partial excision of the cyst wall and drainage of the cavity and subphrenic space. The uninfected cysts were operated using deroofing technique. A year after surgery the patient remained symptom free.

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