Concurrent Intracranial Hypotension and Hypertension in a Patient with Refractory CSF Leak: A Case Report
Pranshi Juneja1, Majd Rachid1, Dechen Samdrup1, Randy D'Amico2, Aaron Zelikovich1
1Department of Neurology, Lenox Hill Hospital, 2Department of Neurosurgery, Lenox Hill Hospital
Objective:
To describe a rare case of concurrent intracranial hypotension and hypertension in a patient with refractory cerebrospinal fluid (CSF) leak that improved with ventriculo-pleural shunt (VPS) placement. 
Background:
Intracranial hypotension and intracranial hypertension are two entities that present as headaches but arise from distinct CSF fluid dynamics. While the former frequently occurs in the setting of leaks, the latter is due to increased pressures. The concurrence of both conditions in a single patient is rare.
Design/Methods:
N/A 
Results:

A 31-year-old woman with Behçet’s disease, gastroparesis, seizures, migraines, and von Willebrand disease presented for persistent headaches after undergoing intrathecal morphine pump placement. She was found to have a CSF leak during surgical intervention. Despite blood patching, fibrin glue, and pump revision, she had persistent positional headaches and development of pseudo-meningocele. Pump removal revealed a macerated dura which was repaired with sutures and sealants. Due to symptom recurrence, a repeat repair with plastic surgery flap closure was attempted and high CSF pressure was noted intraoperatively. Concern for concurrent intracranial hypotension and hypertension prompted VPS placement which led to resolution of the patient’s headaches and lumbar fluid collection. 

Conclusions:

Developing intracranial hypertension following treatment of CSF leaks is rare. The mechanism underlying this “rebound” increased intracranial pressure remains unknown. One possibility is the induction of CSF production in response to prolonged CSF volume depletion. Another possible mechanism is the impaired CSF resorption and a slowing of the bulk flow. Our patient experienced significant debilitation due to low pressure headaches which improved with a ventriculo-pleural shunt placement that typically treats high pressure symptoms. While the patient’s clinical course is most consistent with a paradoxical transition from low to high pressure states, alternate contributors such as aseptic meningitis or Behçet’s-related neuroinflammation should also be considered.

10.1212/WNL.0000000000215826
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