Abstract: Objective　To explore the diagnosis and treatment of disseminated cryptococcosis involving the biliary tract and the central nervous system. Methods　The clinical data of one child with disseminated cryptococcosis involving biliary tract and central nervous system were analyzed retrospectively. The related literatures were reviewed. Results　A 6-year-old boy presented with jaundice, hepatic dysfunction and convulsions. The surgical treatment was performed. The pathological biopsy showed cryptococcal infection, cryptococcal capsular polysaccharide antigen latex agglutination test in serum and cerebrospinal fluid were positive and cerebrospinal fluid ink staining was also positive. After antifungal treatment, the symptoms were relieved and no sequelae existed during the follow-up period. Conclusion　Disseminated cryptococcosis simultaneously involving the biliary tract and the central nervous system is rare in children.