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Paranasal sinus mucocele is a slowly growing benign cystic lesion. It usually involves the frontaland ethmoid sinuses and can extend to adjacent structures, especially to the orbit, skull baseand brain parenchyma. Prompt surgical intervention is needed when symptoms occur. Completeresection of mucocele is approached via endoscopic sinus surgery, while marsupialization isalso widely considered. Recently, we encountered a case of spontaneous brain herniation andcerebrospinal fluid leakage during endoscopic marsupialization of ethmoid sinus mucocele. Herein, we report the case with a review of the literature.