We prospectively evaluated the efficacy and safety of imatinib plus hydroxyurea 
in patients with progressive/recurrent meningioma. A total of 21 patients with 
progressive/recurrent meningioma were enrolled in this dual center, single-arm, 
phase II trial. All patients received 500 mg of hydroxyurea twice a day. 
Imatinib was administered at 400 mg/day for patients not on CYP3A enzyme 
inducing anti-epileptic drugs (EIAEDs) and at 500 mg twice a day for patients on 
EIAEDs. The primary endpoint was progression-free survival at 6 months (PFS-6) 
and secondary endpoints were safety, radiographic response rate, and overall 
survival (OS). Best radiographic response was stable disease and was observed in 
14 patients (67%). PFS-6 for all patients, those with grade I tumors (n = 8) and 
those with grade II or III tumors (n = 13) was 61.9, 87.5 and 46.2%, 
respectively. Patients with grade II or III tumors had poorer PFS and OS than 
those with grade I tumors, (P = 0.025 and P = 0.018) respectively. The only 
grade 3 or greater adverse event occurring in ≥ 10% of patients was anemia 
(10%). Imatinib plus hydroxyurea is well tolerated among patients with 
meningioma but has modest anti-tumor activity for this indication.