Spontaneous intracranial hypotension (SIH) is an uncommon cause of headache which is primarily postural in nature. In extremely rare cases neurological manifestations including cranial nerve palsies, bulbar dysfunction, tremors, ataxia, myelopathy, and cognitive decline can be seen. These atypical presentations can obfuscate the underlying pathology which can result in late diagnosis or misdiagnosis. The objective of this case is to help raise awareness about this condition by describing an uncommon feature that may assist in its early diagnosis and appropriate management.

We report a 41-year-old Hispanic female with history of type II diabetes and hyperlipidemia who presented with 3 weeks of gradually worsening bifrontal positional headache and 5 days of progressive binocular horizontal diplopia and dizziness. Exam notable for isolated left cranial nerve VI palsy.

Preliminary imaging with non-contrast CT head, and CT angiogram were unremarkable. Autoimmune and infectious screens were negative.  MRI with and without contrast of the brain, as well as cervical, thoracic, and lumbar spine demonstrated diffuse pachymeningeal enhancement.  Dynamic CT myelogram revealed CSF leaks at C2 and T10, with extensive extravasation of contrast into cervical paraspinous and interspinous soft tissues.  Patient’s exam remained stable, symptoms gradually improved over week long hospital course, which may be attributed to limited mobility during inpatient workup and some relief with PO caffeine. She was discharged with close neurological follow up and plan for blood patch as an outpatient.

Spontaneous intracranial hypotension is an uncommon condition with its own uncommon presentations.  In the differential of causes of abducens nerve palsies, intracranial hypotension secondary to spontaneous CSF leak should be considered.