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A 49-year-old woman developed symptoms of syringomyelia 3 years after having presented with spontaneous intracranial hypotension (SIH). She had previously undergone two unsuccessful non-targeted epidural blood patches. The MR scan showed features of cervicothoracic syringomyelia and ongoing intracranial features of SIH. Following two further ineffective non-targeted epidural blood patches, a cerebrospinal fluid venous fistula was identified on myelography, which was surgically disconnected. At subsequent follow-up, she showed marked and progressive clinical and radiological improvement. This case shows that offering curative treatment for the underlying cause of SIH is an effective—and the preferred—way of managing SIH-associated syrinx.