Cases of body image pathology are often misdiagnosed. Recognition of the obsessional annoyance with body image, especially with primary and secondary sex characteristics (skoptic syndrome), is important in making a differential diagnosis with other psychiatric and gender identity disorders. We have reported the successful use of lithium carbonate in treating two cases of skoptic syndrome. The initial low dosage (600 mg./day) resulted in dramatic reversal of obsessional thoughts of self-mutilization and self-castration. Increased dosages (900 mg./day) are still well below those usually used for manic-depressive illness. Our choice of lithium carbonate was prompted by our experience in successfully treating other forms of sexually related dysphorias at low doses, its ability to act rapidly, and the minimal side effects. These changes have been dramatic and lasting. In addition, there was marked lessening of dysthymia and intense anhedonia manifested by severe isolation, listlessness, and apathy. We believe that these cases are important to report in order that psychotherapists make better differential diagnoses of psychiatric disorders related to body image pathology and gender identity disorders. We also feel it is important to report a medication which was found effective in reducing the threat of self-mutilating behavior and making patients more receptive to psychotherapy. These cases increase our understanding of sexually obsessive and compulsive drives and some methods which have been found efficacious in reducing these symptoms.