The authors report a case of cerebral venous and sinus thrombosis (CVST) in a patient receiving a low-dose estrogen-progestin combination (oral contraceptives, OCs) for uterine adenomyosis. She was switched to gonadotropin-releasing hormone agonist (GnRHa) draw-back therapy, which was successfully administered long-term. Case: The patient was a 38-year-old nulligravida with a history of smoking. She presented to this hospital with dysmenorrhea and postmenstrual lower abdominal pain. Adenomyosis was diagnosed using ultrasound and magnetic resonance imaging. She was instructed to stop smoking and was administered low-dose OCs. CVST occurred 18 months later. OC therapy was halted, and only antiplatelet therapy was administered. After six months, her chief complaint symptoms intensified, therefore GnRHa draw-back therapy was administered after obtaining informed consent. No uterine enlargement was observed, and the abdominal pain resolved. During 2.5 years of therapy, her bone density levels remained within normal limits. CVST did not recur and no other thromboses were observed.