We report a case of anterior cranial fossa dural arteriovenous fistula (dAVF) in which ocular movement was impaired after Onyx embolization from the ophthalmic artery (OphA). Case Presentation: A 76-year-old male was admitted to our hospital for treatment of an incidentally found anterior cranial fossa dAVF. Onyx was injected from the right anterior ethmoidal artery (AEA) to close the shunt. Onyx refluxed to the third portion of the OphA to make a plug, but was unable to reach the venous side beyond the shunt; therefore, a small shunt remained. Although his visual acuity and field were normal, vertical diplopia developed after embolization and disappeared 1 month later. Diplopia worsened when the patient tilted his head to the right. Neuro-ophthalmological examination confirmed right superior oblique muscle impairment. The cause of diplopia was considered to be ischemic injury of the superior oblique muscle associated with embolization of the AEA, which provides nutrients to the superior oblique muscle and trochlear nerve. Conclusion: Embolization from the OphA beyond the third portion may cause external ophthalmoplegia, although it may heal spontaneously. Keywords▶ dural arteriovenous fistula, complication, Onyx, superior oblique muscle paralysis, anterior cranial fossa dural arteriovenous fistula originally a stapedial artery. 3) When performing embolization through the OphA, it is important to advance a microcatheter to the periphery to the second portion beyond the CRA. It has been considered to be relatively safe to embolize the lesion beyond the third portion of the OphA. 3) Here, we report a case of anterior cranial fossa dAVF showing a rare complication of impairment of extraocular movement developing after transarterial embolization (TAE) with Onyx Liquid Embolic System (Medtronic, Minneapolis, MN, USA) (Onyx) via the anterior ethmoidal artery (AEA). Case Presentation A 76-year-old male was admitted to our department for the treatment of asymptomatic anterior cranial fossa dAVF. The dAVF was incidentally found on magnetic resonance imaging (MRI) performed to evaluate asymptomatic right internal carotid artery (ICA) stenosis. He had medical history of abdominal aortic aneurysm treated by endovascular aortic repair, pulmonary emphysema, dyslipidemia, and angina pectoris. On admission, he had no neurological deficit. This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives International License.