A 49-year-old man had left superior oblique myokymia for eight years. Magnetic resonance images with enhanced spoiled gradient recalled acquisition in the steady state (SPGR) and flow imaging using steady acquisition (FIESTA) disclosed a branch of the superior cerebellar artery lying on the root exit zone of the left trochlear nerve. Posterior fossa craniotomy confirmed the imaging findings. A Teflon pad was placed between the compressing artery and the trochlear nerve. The patient's superior oblique myokymia has completely resolved with a one-year follow-up. Only one such case has been previously reported. This is the first report to display the imaging findings.