The authors present the case of a 55-year-old man suffering from intractable spontaneous intracranial hypotension, in whom conservative treatment with 19 weeks of bed rest was not effective. In this period the patient twice underwent surgery for bilateral chronic subdural hematoma, a complication of spontaneous intracranial hypotension. Conventional radionuclide cisternography, magnetic resonance imaging, and computerized tomography myelography did not demonstrate cerebrospinal fluid (CSF) leakage. Repeated radionuclide cisternography with the patient in an upright position revealed leakage of the tracer at upper cervical levels. Computerized tomography myelography with breath holding also showed CSF leakage of the contrast medium bilaterally at upper cervical levels. The patient underwent surgery, and bilateral C-2 and C-3 spinal nerve root pouches were sealed off from the subarachnoid space with oxidized cellulose cotton and fibrin glue. Epiarachnoid spaces around the root sleeves were also sealed to ensure complete resolution of the CSF leakage. After the surgery, the patient was completely free of the disease. In the case of intractable persistent spontaneous intracranial hypotension, surgical treatment is preferable to long-term conservative management. To identify CSF leakage, radionuclide cisternography with the patient in the upright position is useful. When obvious leakage is encountered, surgical sealing of the lesion should be performed via a subarachnoid approach.