Trichosporon spp. are yeast-like microorganisms responsible for skin, urinary, pulmonary, or bloodstream infections. Due to intrinsic resistance to echinocandins, poor susceptibility to polyenes, and preferred occurrence in immunocompromised patients, such infections are often of poor prognosis. Yet no consensual therapeutic guidelines are presently available. Several clinical cases of Trichosporon infections have been successfully treated with azole therapy, including voriconazole which appeared frequently effective against Trichosporon both in vitro and in vivo. However, the low efficacy associated with some Trichosporon genotypes, complex pharmacokinetics, and the side effects of voriconazole represent limitations for its use and has prompted a search for other therapeutic options. Here, we report a case of T. asahii fungemia in a patient with B-cell acute lymphoblastic leukemia which was successfully treated with isavuconazole consecutive to stopping voriconazole therapy due to severe side effects. This observation suggests that isavuconazole with a similar spectrum to voriconazole, fewer pharmacology interactions, and side effects may be considered as a valuable therapeutic option against Trichosporon infections.