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Vagus nerve stimulation (VNS) is an effective therapeutic option that is widely used worldwide in drug-resistant epilepsy cases. Because it is a surgical procedure, some complications may develop with VNS implantation. Although VNS-related pain symptoms have been reported, VNS-related trigeminal neuralgia is an unexpected and rather rare side effect. This report presents a case of trigeminal pain as an adverse effect of VNS. A patient with drug-resistant epilepsy undergoing VNS treatment developed pain synchronously with stimulation in his left upper and lower jaw and teeth. Pain occurred on the day of stimulation’s current intensity (SCI) increase. The sudden disappearance of pain with decreasing SCI suggested that trigeminal pain was related to VNS. Because it is a rare side effect, trigeminal pain may not be regarded as a VNS-related side effect and may lead to unnecessary examinations. Being a rapidly reversible side effect, recognizing it and reducing SCI is crucial. VNS stimulation paradigms on nociception are still largely unknown, and it will be an important step to elucidate the important impact of VNS in pain modulation.