We present a case of spontaneous regression of multicentric pilocytic astrocytoma with cerebrospinal fluid (CSF) dissemination without neurofibromatosis type 1 (NF1) in an adult, the first such case reported. Magnetic resonance imaging (MRI) showed multiple low signal intensity lesions on T1-weighted images and high signal intensity areas on T2-weighted images in the bilateral thalamus, basal ganglia and midbrain. Contrast-enhanced MRI revealed that small, enhanced lesions were seen in the basal ganglia and the pineal region. Neuroendoscopic biopsy and third ventriculostomy were performed. Intraoperative findings demonstrated CSF dissemination. Histologically, the specimens showed pilocytic astrocytoma. Serial MRIs showed regression of the tumor without any additional treatment. The clinical features of spontaneous regression of pilocytic astrocytoma are discussed.