In this population-based study, we determined the frequency and clinical characteristics of leptomeningeal disease (LMD) developing in the context of oligodendroglial tumors (oligodendrogliomas and oligoastrocytomas). LMD occurred in only 3.9% (8/204) of oligodendroglial tumors and in patients with more recurrences [mean 2.88 vs. 1.27 in LMD and non-LMD, respectively (p = 0.001)]. In contrast to LMD from systemic solid tumors, the median survival following the diagnosis of LMD in oligodendroglial tumors was surprisingly long at 22 months (95% CI 11-33 months). Treatment with oral chemotherapy seemed as effective as more aggressive treatments (e.g. repeat RT or intrathecal chemotherapy) in these patients.