BackgroundEsthesioneuroblastomas (ENBs) are rare, aggressive tumors arising from the olfactory neuroepithelium in the superior nasal tract. Current treatment strategies typically involve a combination of surgery and radiation, with systemic chemotherapy considered on a case-by-case basis. However, the role of intra-arterial (IA) therapies for ENBs remains underexplored. In this report, we present an illustrative case from our center and a systematic literature review compiling an overview of similar cases.MethodsThe case of an elderly man with recurrent ENB treated with IA carboplatin is described. We systematically reviewed PubMed and Embase to gather data on the indications, treatment course, and outcomes of IA chemotherapy for ENBs.ResultsThe patient presented initially with epistaxis and was diagnosed with ENB post-resection and radiation. Recurrence occurred 28 years later, causing visual loss and headaches. Multiple sessions of stereotactic radiosurgery over 2 years were ineffective. After multispecialty discussions, IA carboplatin was selected for neoadjuvant therapy. The bilateral internal maxillary arteries (IMAs) were identified as primary feeders of the recurrence. Carboplatin was directly administered via the IMAs in two stages, 1 month apart. The procedures were well-tolerated, and the patient was discharged without complications. Despite symptom improvement with radiographically stable tumor at 2-month follow-up, the patient opted for palliative care. The systematic review identified four ENB cases involving the ethmoid sinus with varying extensions into anterior and middle cranial fossae. IA chemotherapeutics were administered through maxillary or carotid arteries as an adjunct to resection, radiotherapy, or systemic chemotherapy, with no intraprocedural or postprocedural complications reported. Two patients died during follow-up: one with primary ENB and another with recurrent ENB.ConclusionThe safe administration of IA chemotherapeutics for primary and recurrent ENB in this case and literature review suggests a potential role for IA therapies in managing intracranial tumors, especially with advanced superselective microcatheter techniques.