Objective: To report a rare case of true hermaphroditism presenting in adolescence as bilateral gynecomastia in a phenotypic male.

Methods: Case report. Methods: Academic medical center. Methods: A 19-year-old phenotypic male who presented with pronounced bilateral gynecomastia and was ultimately diagnosed with true hermaphroditism. Methods: Endoscopic removal of the internal female genital tract (hemiuterus and ovary), reduction mammoplasty, repair of penile chordee, and left testicular prosthesis placement. Methods: Diagnosis and treatment of true hermaphroditism.

Results: Bilateral mammoplasties, repair of a penile chordee, placement of a left testicular prosthesis, and excision of the left hemiuterus and ovary resulted in return to testicular function in this true hermaphrodite.

Conclusions: Although rare, true hermaphroditism should be suspected in male patients presenting with bilateral breast enlargement in adolescence.