Tumors of the central nervous system in fish are rare, and only six cases of spontaneous olfactory neuroepithelioma have been reported. This is the seventh case, found in a medaka, Oryzias latipes. The tumor was noted near the right olfactory orifice and finally measured 1.5 mm in diameter. Histologically the tumor consisted of undifferentiated neuroblasts forming a few true rosettes. Mitosis was frequently observed. Tumor cells stained diffusely for neuron-specific enolase and sporadically for neurofilament proteins by immunohistochemical procedures. Additionally a few large tumor cells were positively stained for S-100 protein. Electron microscopy revealed that the tumor cells had extended cytoplasm in which parallel neurotubules and a few neuroendocrine granules were noted. In the perinuclear region, bundles of intermediate filaments and neuroendocrine granules were seen. Single cilia and a pair of centrioles were occasionally found, but no ciliated cells were found in this tumor. Some large tumor cells contained electron-dense intracytoplasmic inclusions which showed a crystalloid structure by high-magnification electron microscopy; however, this type of crystalloid has never been reported in neuronal tumors.