The rare occurrence of an inflammatory mucocele in a pneumatized anterior clinoid process is described. The patient, a 20-year-old woman, presented with a severe visual field defect in her right eye associated with abnormality in the right anterior clinoid process identified on computed tomography and magnetic resonance imaging. Initially, surgical intervention was recommended for resection of a possible neoplasm. The patient's clinical history, however, was significant for sinusitis accompanied by sore throat and right ear infection that resolved with oral antibiotic therapy. When her condition was evaluated approximately 1 month after the onset of her visual symptoms, the patient had regained full visual acuity in the affected eye. Surgical exploration was not required, and the patient's optic neuropathy reversed with appropriate antibiotic therapy.