A 33-year-old woman who presented with vaginal bleeding was diagnosed to have neuroendocrine small cell carcinoma based on cervical smear and biopsy. Hysterectomy was performed, and a tumor measuring 5.5 X 2 mm was found at the squamocolumnar junction of the uterine cervix. In the immediate vicinity of the tumor, there was proliferation of cytologically benign endocrine cells in the normal endocervical glands and in the glands showing intraepithelial glandular neoplasia. Both the hyperplastic endocrine cells and the invasive tumor cells showed argyrophilia and immunostaining for neuron-specific enolase, neurofilament, and chromogranin. The topographical relationship suggests that endocrine cell hyperplasia may represent a precursor of neuroendocrine carcinoma of the cervix.