Introduction:: Bilateral testicular regression syndrome (TRS) is a very rare condition characterized by the absence of testicular tissue in a genotypic male.
Case report:: A 39 years old male with TRS (diagnosed at age 8, during investigation for bilateral cryptorchidism) associating paranoid schizophrenia (diagnosed at the age of 14) is described. The patient received parenteral androgen replacement only for a few months in the pubertal period. After puberty, the psychiatric team strongly advised against the androgen replacement which was stopped and not resumed ever since. At presentation the patient has no subjective complaints but clear signs of hypogonadism. The psychiatric status was stable under neuroleptics. Topical androgen replacement was recommended but it aggravated the psychotic aggressive features so it was stopped.
Conclusions:: We present the case of a patient with TRS associating, from early adolescence, a severe psychiatric pathology with aggressive characteristics, which constantly precluded adequate testosterone replacement.