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To investigate the ultrastructural and clinical characteristics of melanoma of the central nervous system (CNS). The clinical and electron microscopy pathology data of nine patients with melanoma surveyed from 1993 to 2017 were analyzed. All the CNS melanomas were confirmed by transmission electron microscopy (TEM), including eight cases of primary melanomas and one case of metastatic melanoma. In this study, four stage II melanosomes were intracranial space-occupying, three of which were malignant melanoma, the other one was melanoma. Among the five stage IV melanosomes, four cases were intraspinal space-occupying, the other one was intracranial space-occupying, and the pathological diagnoses were all melanoma. At present, TEM is an important tool for the diagnosis of CNS melanomas. Malignant melanoma has high malignancy and recurrence rate and poor prognosis, while benign melanoma with relatively low recurrence rate, so we speculate that patients with mainly immature melanosomes are more likely to exhibit recurrence.