We report the case of a 22-year-old woman who developed multicentric pleomorphic xanthoastrocytomas (PXAs) in the cerebral hemisphere. She underwent a first operation for a PXA in the right parietal lobe at the age of 7 years and a second operation at the age of 15 years in the right frontal lobe, remote from the previous tumor site. At age 22 years, she was found to have a tumor, which was a newly formed PXA, in the left occipital lobe. There was no recurrent tumor in the right frontal lobe. Clinical and pathological aspects of multicentric PXAs are reviewed and discussed.