Simple liver cysts occasionally cause pressure symptoms of the abdomen. We herein report an extremely rare case of spontaneous rupture of simple liver cysts. A 65-year-old woman suffered abdominal fullness and dyspnea. Laboratory examinations revealed general inflammation and mild hepatorenal dysfunction. Computed tomography revealed giant polycystic liver and ascites. Echinococcus antibody was not detected. Abdominal paracentesis provided dark brown transparent ascites in which any parasites or tumor cells were not observed. We diagnosed spontaneous rupture of isolated polycystic liver disease (PCLD) and continuously drained the ascites. After the symptoms and laboratory data were improved, resection of liver cysts and left lateral segmentectomy were performed. Histopathologically, simple columnar epithelia inside of cyst walls were observed. The patient remains well without recurrence of the symptoms 10?months after the surgery. We reviewed characteristics of PCLD and considered appropriate treatment for spontaneous rupture of simple liver cysts based on the previous case reports including the present case
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