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Objective: Cysts of the septum pellucidum (CSPs) may become symptomatic because of obstruction of cerebrospinal fluid flow, resulting in increased intracranial pressure and hydrocephalus requiring surgical intervention. Endoscopic fenestration may be the most effective and least invasive technique to treat this pathological condition., Clinical Presentation: An 11-year-old boy sought treatment for frequent episodes of severe headache. On examination, he had papilledema. There was evidence on magnetic resonance imaging scans of a space-occupying CSP with obstructive hydrocephalus., Intervention: The endoscopic technique of fenestration of both lateral walls of an enlarged CSP via a left frontal approach under ultrasound guidance using a rigid endoscope was successful. After surgery, the patient became asymptomatic, his papilledema resolved, and magnetic resonance imaging scans demonstrated collapse of the walls of the CSP toward the midline., Conclusion: Neuroendoscopic fenestration should be strongly considered as the treatment of choice for symptomatic CSPs. This procedure alone can lead to complete resolution of clinical symptoms and hydrocephalus, can reduce the size of the CSP, and can obviate the need for an implantable cerebrospinal fluid shunt.