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The aim of this study was to review a series of patients submitted to hepatectomy for primary intrahepatic lithiasis to evaluate early and late results with an assessment of indications, methods and long-term outcomes. From January 1992 to December 2007, 40 patients (25 males and 15 females with a mean age of 51 years) underwent surgery for primary intrahepatic lithiasis in our Hepato-biliary Surgery Unit. Left hepatectomy (20 patients) and left lateral segmentectomy (12 patients) were the most common procedures performed. A cholangiocarcinoma was found in 4 patients (10%) and only two of these underwent liver resection, while an exploratory laparotomy was performed in the remaining two patients for an unresectable tumour, unexpected before surgery. There was no postoperative mortality. The morbidity rate was 22.5% with a prevalence of infectious complications related to bile leakage. Long-term results, assessed in 30 patients with a follow-up longer than 12 months, were good or fair in 28 patients (93.3%). Primary intrahepatic lithiasis is diagnosed increasingly in Western countries as a result of the improvement in imaging techniques. The stones originate inside the liver at the level of dilatations of the bile ducts above congenital strictures of the main hilar ducts. Biliary pain and cholangitis are the most common presenting symptoms, whereas cholangiocarcinoma represents the unfavourable complication of the disease. In the majority of cases, a single liver lobe or segment is involved and liver resection allows definitive treatment of the disease and prevention of cancer.