To demonstrate spontaneous regression of large, clinically symptomatic optic pathway gliomas in patients with and without neurofibromatosis type 1 (NF-1). Methods: Patient cases were collected through surveys at 2 consecutive annual meetings of the North American Neuro-Ophthalmology Society (NANOS) and through requests on the NANOSNET Internet listserv. Serial documentation of tumor signal and size, using magnetic resonance imaging in 11 patients and computed tomography in 2 patients, was used to evaluate clinically symptomatic optic pathway gliomas. All tumors met radiologic criteria for the diagnosis of glioma and 4 patients had biopsy confirmation of their tumors. In 3 patients, some attempt at therapy had been made many years before regression occurred. In one of these, radiation treatment had been given 19 years before tumor regression, while in another, chemotherapy had been administered 5 years before signal changes in the tumor. In the third patient, minimal surgical debulking was performed 1 year before the tumor began to shrink. REPORT OF CASES CASE 1 A healthy 5-year-old boy was believed to have severe amblyopia with hand motion vision in the right eye during a school examination. At age 7 years, right optic disc atrophy was recognized. His left eye was normal. He had 7 café-au-lait spots, but no Lisch nodules on his irides. There was no history of NF-1 or ocular problems in his family. Magnetic resonance CLINICAL SCIENCES Author affiliations are located at the end of this article.

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