A 61-year-old man was admitted to the emergency room complaining of a severe left exophthalmos caused by frontal and ethmoid sinus mucoceles that were visualized on a brain computerized tomogram. In addition, he had coexisting ankylosing spondylitis with a 20 year duration that resulted in total fixation of the cervical spine and progressive thoracic kyphosis. An unruptured anterior communicating artery aneurysm was found incidentally on the cerebral angiogram. We report that the anesthetic management for endoscopic sinus surgery of a frontal sinus mucocele in a patient with coexisting severe cervical spine ankylosing spondylitis and an unruptured cerebral aneurysm requires a detailed preoperative assessment of the airway, cardiac, pulmonary, and neurologic system. This case highlights the need for careful measures to avoid rupturing the cerebral aneurysm by the increased blood pressure induced by endotracheal intubation and the infiltration of an epinephrine-containing local anesthetic.