BACKGROUND: This study reports the combined experience of the University of Miami and University of Pittsburgh with endoscopic endonasal resection of esthesioneuroblastoma (ENB). A retrospective case series review was performed in a tertiary care university hospital. METHODS: Twenty-three patients, 16 men and 7 women, were reviewed. Mean age was 56.6 years (15-79 years). Nineteen patients received primary endoscopic endonasal anterior skull base resection. Of these, the modified Kadish stage at presentation was A in 2 patients, B in 11 patients, C in 5 patients, and D in 1 patient. Three patients had revision surgeries for recurrent tumors. The main outcome measures were complete resection and margin assessment, short-term and long-term complications, and recurrence rate. RESULTS: Complete resection and negative intraoperative resection margins were achieved endoscopically in 17 of the primarily treated cases. The two other cases had one patient that required an additional craniotomy approach to complete the resection of a positive lateral dual margin, another patient had positive margins at the orbital apex. All patients tolerated the endoscopic procedure very well with no meningitis. There were four cerebral spinal fluid leaks. Mean follow-up period for the primarily treated cases was 45.2 months (11-152 months), all were disease free at the most recent available follow-up. CONCLUSION: In experienced hands and carefully selected patients, endoscopic resection of ENB respects the principles of oncologic surgery, providing an adequate exposure for margin assessment as well as reliable reconstruction of the anterior skull defect with a relatively low morbidity.