The impact of hypogonadism on the probability of retrieving testicular sperm from patients with non-obstructive azoospermia (NOA) is still a matter of debate. Conflicting evidence in this field may be justified by the striking differences between serum and intratesticular testosterone (ITT) levels found in men with severe spermatogenic dysfunction, so that normal ITT levels may coexist with low serum testosterone levels. Here we report the case of a patient with NOA with a steadily reduced serum testosterone level irresponsive to hormonal stimulation with human chorionic gonadotropin. Supported by his normal serum 17-hydroxyprogesterone (17 OHP) levels, previously suggested to be marker of ITT levels, microdissection testicular sperm extraction was performed for both testes on two separate occasions, resulting in the retrieval of enough sperm for ICSI. Three ICSI cycles were then performed, one blastocyst was transferred, and five were cryopreserved. This case report suggests that normal serum 17 OHP levels, being suggestive of normal ITT levels, may support the decision to proceed with surgical sperm retrieval in hypogonadal patients with NOA, even for those irresponsive to hormonal treatment.