Cough headache may be the clinical manifestation, sometimes isolated, of an intracranial disease. There are several possible causes of secondary cough headache. The hypothesis that cough headache may be the expression of spontaneous intracranial hypotension has been advanced only recently. In fact, this would represent an exception to the rule that cough headache is generally secondary to conditions leading to an increase in intracranial pressure and/or volume. We report and discuss a case of cough headache secondary to spontaneous intracranial hypotension in an otherwise healthy 59-year-old man. The condition was complicated by cerebral venous thrombosis.