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In the diagnosis of an intracranial dural arteriovenous fistula (DAVF), arterial spin labeling (ASL), a sequence of magnetic resonance imaging (MRI) to depict high-blood-flow intracranial lesions, has been reported as a useful and noninvasive tool, not only to predict the presence of cortical venous drainage and draining veins, but also to confirm persistent obliteration after treatment. However, such utility of ASL has not been reported in DAVF of the craniocervical junction (CCJDAVF) because of the rarity of this disease and uncertainty in the acquisition of precise images.We report a case of CCJDAVF presenting with myelopathy. Preoperative ASL images showed an abnormal high-intensity signal in the craniocervical junction, consistent with the anterior spinal vein and draining veins, which were also identified by digital subtraction angiography. After successful surgical treatment for the disease, MRI and 4-dimensional computed tomography angiography (4DCTA) confirmed complete disappearance of CCJDAVF. The ASL images also showed no abnormal intensity signal. The patient was followed-up using ASL, and no recurrence of high-intensity signal was observed.As repetitive image examination is mandatory in the follow-up of a patient with DAVF to exclude recurrence, ASL is highly beneficial because of the unnecessity of an exogenous contrast medium and high credibility to depict the disease. The craniocervical junction may be out of the field of view in routine MRI. Special attention must be paid to setting the field of view and post labeling delay (PLD) to obtain precise images of ASL in CCJDAVF.