The histologic, immunocytologic, and electron microscopic features of pituitary adenomas surgically removed from two men with elevated levels of blood follicle-stimulating hormone (FSH) are described. In both cases, the high blood FSH levels were reduced after surgery. By light microscopy, the tumors corresponded to chromophobic adenomas, and the immunoperoxidase technique revealed the presence of immunoreactive FSH (beta-subunit) in the cytoplasm of the adenoma cells. By electron microscopy, the adenoma cells differed considerably from nontumorous FSH cells. They were smaller and angular and contained numerous microtubules as well as spherical secretory granules measuring 100 to 250 nm in diameter and often lining up along the cell membranes. The present findings are consistent with the view that FSH-producing adenomas may originate in the human pituitary.