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OBJECTIVE Sylvian fissure (SF) arteriovenous malformations (AVMs) are among the most challenging vascular lesions amenable to neurosurgical treatment and account for 10% of all locations. As radiosurgery and endovascular techniques are increasingly involved in multimodal management protocols, the role of microsurgery needs to be reassessed as a stand-alone technique. The aim of this study was to show that total excision can be achieved with reasonable levels of morbidity and mortality in a real-world setting from a specialized high-volume center. METHODS Forty-three patients with SF AVMs were identified from a series of 577 AVM patients treated microsurgically over a 22-year period. The mean patient age was 33.07 years (range 15–60 years), and there were 22 male and 21 female patients. The mode of presentation was headache in 51.2%, hemorrhage in 34.9%, seizures in 30.2%, and steal phenomenon in 9.3%. The authors analyzed the anatomical basis and angiographic characteristics of such lesions. RESULTS In the preoperative period, 83.7% of the patients had a modified Rankin Scale (mRS) score of 0–2, and 16.3% had an mRS score of 3–5. After a 12-month follow-up, 95.3% of patients had an mRS score of 0–2, and 4.7% had a score of 3–6. The difference between pre- and postoperative scores was not statistically significant. SF AVMs have several particular features: 1) They produce angiographic steal of the anterior cerebral artery. 2) The nidus is fed by only one of the main trunks of the middle cerebral artery (MCA). 3) Participation of deep perforators is uncommon. 4) They have two or more early draining veins showing their fistulous nature. 5) Preoperative embolization and radiosurgery have a low rate of permanent cure. CONCLUSIONS These AVMs represent a surgical challenge due to their proximity to critical structures such as the MCA, insula, internal capsule, and speech and memory functions in the dominant hemisphere. Essential key points are the wide opening of the SF and proper differentiation between feeders and normal vessels. Although this location can seem daunting, SF AVMs carry no additional surgical risk if adequately managed.