Spontaneous intracranial hypotension is characterized by orthostatic headache caused by cerebrospinal fluid (CSF) leakage. However, clinical presentation of SIH is variable, and normal or high intracranial pressure in CSF study is not uncommon. Infratentorial superficial siderosis (ISS) shares similar pathomechanism with SIH, as developed after chronic CSF leakage, whilst several years of latency after SIH onset. Here, we report a 47-year-old male patient who had experienced prominent orthostatic headache twenty years before, and presented reverse-orthostatic headache in this time, accompanying with radiological features of SIH and ISS in brain magnetic resonance imaging (MRI). CSF leakage was confirmed by spinal MRI and MR myelography, and his headache was aggravated after epidural blood patch.