ObjectiveTo summarize the clinical features of an adult patient with occult cerebral alveolar echinococcosis with liver and lung infection. MethodsA Tibetan male patient in his middle age from the epidemic area of echinococcosis infection was diagnosed to have liver, lung and cerebral alveolar echinococcosis infection in Ganzi People's Hospital. He had the resection surgery, and the pathological result confirmed the primary diagnosis. We searched the literatures from January 1985 to December 2015 for occult cerebral alveolar echinococcosis and reviewed all the full texts in China Journal Full-text Database. Seventeen articles were qualified and 42 patients were reported. Combining with the relevant English literature using Medline, we analyzed the epidemic, pathophysiological and clinical manifestations of cerebral alveolar echinococcosis infection and explored the methods of prevention and treatment. ResultsAccording to the results of literature analysis, cerebral alveolar echinococcosis appeared often secondary to infection of other organs. Nervous system symptom concealed or progressed slowly; imaging and pathological tests were important for diagnosis. Resection surgery was the essential method of cure. ConclusionAlveolar echinococcosis can affect multiple organs. In patients without neurological symptoms, if other organs are found to be infected, it is important to screen patients with intracranial involvement. Because this kind of patients with intracranial lesions with hydatid are often secondary to other organ infection, active treatment in early phase is necessary in order to avoid further expansion of lesions and metastasis.