Twenty-one patients for whom adequate clinical data were available were identified in a retrospective review of cases of Candida species isolated from cerebrospinal fluid (CSF) following neurosurgery; 86% had indwelling cerebrospinal devices (shunts). Candida species were isolated from multiple CSF samples from 10 patients; CSF samples from seven of 10 were initially drawn through indwelling devices and those from nine of 10 were obtained by subsequent lumbar punctures. All of these patients were treated with antifungals, although therapy was delayed in 50% of cases until the second positive culture was reported. In 11 cases, Candida was the only isolate recovered from CSF samples drawn through indwelling devices; cultures of subsequent CSF samples obtained by lumbar puncture were negative in 10 of 11 cases. Only two patients for whom a single culture was positive for Candida species were treated with antifungals (both of whom were symptomatic), and none of the untreated patients died of infection. The clinical significance of a single positive CSF sample drawn through an indwelling device is difficult to assess, and a definitive diagnosis may require repeated cultures of CSF samples obtained by lumbar puncture.