Objective: We report a single case of resolution of chronic incapacitating headache following surgical fenestration of a large pineal cyst.
Background: There is an established link between pineal cysts and headaches particularly in the presence of ventriculomegaly and gaze paresis. Our patient’s chronic headache was attributed to a large pineal cyst.
Result: Following surgical fenestration there was immediate, complete relief of the patient’s unremitting headache. Ten months since the operation the patient remains headache free.
Conclusion: This case demonstrates that surgical intervention may play a role in management of patients who have large benign pineal cysts and intractable headache despite the absence of ventriculomegaly and gaze paresis.

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