ABSTRACT A 58 year old male with anosmia and primary testicular failure is described. The presence of anosmia did not preclude the secretion of elevated levels of gonadotrophin suggesting that in hypogonadotrophic hypogonadism with anosmia (Kallmann's syndrome) the hypogonadotrophism is not the result of anosmia alone, but that other, probably hypothalamic, factors are involved. The patient described phenotypically resembled Klinefelter's syndrome, but no chromosomal abnormality was detected. To our knowledge, hypergonadotrophic hypogonadism associated with anosmia has not previously been reported, and thus may represent a heretofore unrecognized clinical entity.