The combination of trigeminal neuralgia and ipsilateral hemifacial spasm, known as painful tic convulsif (PTC), is a relatively rare entity in neurovascular compression syndrome. A case of PTC attributable to different offending arteries is described, the mechanisms and characteristics of PTC are discussed, and a review of the literature is presented. This 80-year-old woman had a 10-year history of left trigeminal neuralgia and ipsilateral hemifacial spasm. She presented with intermittent left facial twitching and pain, especially upon swallowing. MRI revealed compression of the left trigeminal nerve by the left anterior inferior cerebellar artery and of the ipsilateral facial nerve by the posterior inferior cerebellar artery. Microvascular decompression of the lesions via left lateral suboccipital craniotomy resulted in immediate and complete symptom improvement. Our case demonstrates that different arteries can affect the trigeminal and facial nerve at a stage that precedes compression by a tortuous vertebrobasilar artery. We suggest that the presence of PTC should be considered in patients with a tortuous vertebrobasilar artery, irrespective of the offending arteries.