The aim of this report is to investigate the pathogenesis and surgical treatment of encephalocele located in the sphenoid sinus and presented with spontaneous rhinorrhea. The data of a patient with sphenoid sinus encephalocele was analyzed in association with the review of literature. The patient admitted to our clinic with rhinorrhea from the left nasal cavity. Cranial magnetic resonance imaging revealed bone defects in the left sphenoid sinus wall, and the presence of brain tissue and cerebrospinal fluid (CSF) in the sphenoid sinus. The patient underwent dural repair via left pterional approach. Rhinorrhea disappeared after surgery. The pathogenesis of the encephalocele in the sphenoid sinus is not clear. It may be related to the presence of lateral craniopharyngeal canal, extra-gasification in the sphenoid sinus, high intracranial pressure or other factors. It is very important to diagnose accurately the source of CSF fistula in preoperative period, choose the specific operation technique, and follow-up the patient for a long time period.
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