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Background: Spontaneous intracranial hypotension (SIH) is an uncommon, benign, and generally self-limiting condition caused by low cerebrospinal fluid (CSF) volume and pressure usually caused by a CSF leak. Patients with SIH have an increased incidence of subdural hematomas (SDH), which may be bilateral and recurrent. Case Description: We report a unique case of a man presenting with SIH and bilateral SDH that were drained with bilateral craniotomies. During drain removal, the patient had an acute neurological deterioration and a CT scan showed SDH recurrence. The patient had two new recurrent SDH afterwards. After the third surgical intervention, the drain was removed in the OR with concomitant subdural saline infusion, there was no recurrence of SDH after that and the patient has had no further complications after a 2-year follow-up. Conclusion: Patients with intracranial hypotension are predisposed to form SDH. In this case, drain removal caused further decrease in intracranial pressure and triggered a new SDH formation, subdural saline irrigation masked atmospheric pressure and prevented this complication from happening again.

Acute subdural hematoma recurrence during drain removal associated with spontaneous intracranial hypotension – A non-reported complication