Juvenile angiofibroma (JA) is a highly vascularized tumor that often recurs or regrows. Recently, gamma knife surgery (GKS) was attempted on unresectable or remnant JA. We experienced a JA case that developed cerebrospinal fluid (CSF) rhinorrhea 4 years after GKS. Two surgical excisions using midfacial degloving approach were performed before GKS. After radiosurgery, the tumor was controlled, and no early complications were observed. However, 4 years after, intractable CSF leakage developed as a late complication, and we thus performed 4 subsequent endoscopic surgical repairs. The CSF leakage is very rare but can occur as a late complication of GKS and should be treated aggressively.
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