There are currently 13 indications approved by the U.S. Food and Drug Administration for use of hyperbaric oxygen therapy. The European Consensus Conference on Hyperbaric Medicine has 28 indications approved for its use. However, neither includes the use of hyperbaric oxygen therapy for neurological conditions such as migraines with aura. Recent research has made the attempt to fully understand the use of hyperbaric therapy in treatment of neurological conditions, but results have so far been inconclusive. We report a 23-year-old female with an 11-year history of migraines with aura who has received inadequate pharmacological treatment for her migraines since she began having them. Migraines have led her to significant loss of function. The patient underwent treatment at 1.5 absolute atmospheres in a hyperbaric chamber Monday through Friday for 1 hour each day for a total of 40 sessions but reported missing a few sessions over the 8-week period. No more than 1 session during a given week was missed and the patient received no other treatments for her migraines throughout this time period. By her 24th treatment, the patient had only experienced a single migraine with aura but without debilitating pain. The patient stated she had never had a migraine with such little intensity prior to initiation of hyperbaric treatment and did not have to take any days off from work or school. Follow-up at the end of her 40-day treatment period revealed a highly-satisfied patient who had only experienced the single episode of a mild migraine during the entire course of treatment. Thus, we believe that further research needs to be done to realize the full potential of hyperbaric oxygen therapy in the treatment of neurological conditions as this case highlights the potential for using hyperbaric oxygen therapy as prophylaxis against attacks in patients with treatment resistant migraines with aura.