Background <p>Gender dysphoric delusions are reported in about 20–25% of patients with schizophrenia. There have been no previous cases of psychotropics contributing to the occurrence of gender dysphoric delusions or cases where two different types of gender dysphoric delusions occur in the same patient at different times. </p> Case presentation <p>We report on a 30-year-old female with bipolar disorder who after being maintained on sodium valproate and once-monthly paliperidone, experienced side effects in the form of hirsutism (facial hair) and amenorrhea. After discontinuing them, she started reporting that her lower half was female and her upper half was male, then began to identify as “a man trapped in a woman’s body” and expressed a desire to undergo gender affirming surgery. Treatment with a clozapine resulted in her identifying once again as 100% female.</p> Conclusions <p>This is the first case to report on the contribution of psychotropics in the development of gender dysphoric delusions and is also the first to report on two different types of gender dysphoric delusions occurring in the same patient at different times, with initial delusions of simultaneously belonging to both genders and then delusions of belonging to the opposite gender only.</p>