Testicular leiomyomas and gonadal dysfunctions are not commonly reported in teleost and particularly in broodstock fish. In the present work, a testicular leiomyoma related to an unusual case of spermatogenic failure syndrome, in a broodstock seabass coming from an Italian aquaculture farm, is reported. At gross pathology, the circumscribed neoplasia showed several white nodular masses, originating from the gonad, deforming the anatomo‐physiological shape. From light microscopy, the presence of neoplastic tissue islands with spindle cells with a whirling pattern and the low presence of connective tissue were evident and originated from smooth muscle cells. Seminiferous tubules showed severe azoospermia with the absence of sperm cells even closer to the neoplastic area. Immunohistochemical analysis was positive for alpha‐SMA, desmin and vimentin but negative for S‐100, confirming a diagnosis of leiomyoma. This is the first report of spermatogenic failure syndrome associated with a testicular leiomyoma in fish from broodstock. More efforts should be made in studying broodstock fish pathologies related to fish maintenance and hormonal treatments that could economically affect aquaculture production.