BACKGROUND: Diagnosis and treatment of neurocysticercosis have always been a challenge, especially in Vietnam, a resource-restricted environment. Here, we reported successful surgical management of cerebral peduncle neurocysticercosis. PRESENTATION OF CASE: A 65-year-old man with no medical history complained of headache and progressive right-sided hemiparesis for one month prior to admission. On examination, his muscle strength was 3/5 (Frankel grade) with positive Hoffmann and Babinski's signs on the right side. His brain's magnetic resonance imaging suggested a cystic brain tumor (29 x 18 mm) situated in the left cerebral peduncle and hypothalamus region. Enzyme-linked immunoassays (ELISA) of serum for Cysticercus cellulosae, Echinococcus, Toxocara, and Amebiasis were negative. The operation was performed with the modified frontotemporal-orbitozygomatic approach. We extirpated both the cysts. Histopathological examination revealed the larva of Taenia solium. Postoperatively, the hemiplegia was completely recovered, and no complication was reported DISCUSSION: The most recent revised diagnostic criteria of neurocysticercosis emphasised that neuroimaging studies play the most crucial role while clinical manifestations and exposure criteria were only indirect proofs of neurocysticercosis. Neuroimaging and clinical evidence in this patient, however, were more suggestive of cystic brain tumor, which was our most likely preoperative diagnosis. Consequently, total tumor resection required for both histopathological examination and alleviating his hemiplegia. CONCLUSIONS: Preoperative diagnosis of neurocysticercosis has always been a challenge. Total resection of the cerebral peduncle neurocysticercosis was adequate when the patient had localized neurological deficits. (C) 2020 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY-NC-ND license (http://creative commons.org/licenses/by-nc-nd/4.0/).