A 1 yr old castrated male American Staffordshire terrier mix was treated for Scopulariopsis brevicaulis that was discovered during an evaluation of chronic rhinosinusitis. A sinusotomy was performed, with intraoperative infusion of amikacin-impregnated gel and systemic administration of postoperative amikacin. Antibiotic therapy was initially chosen because of historic culture of Mycoplasma. Fungal culture of the frontal sinus identified S. brevicaulis by combined phenotypic characterization and DNA sequencing after morphologic analysis failed to identify the organism. Systemic terbinafine therapy was instituted after fungal culture results were available. The patient recovered well and has had no clinical relapse of disease 3 yr after treatment. This is the first reported case of a fungal rhinosinusitis caused by S. brevicaulis in a dog. Reports of Scopulariopsis infections in humans indicate that treatment of S. brevicaulis infection is hampered by multidrug resistance; prognosis may be poor in people with invasive infections and is currently unknown in dogs. Multimodal therapy, including concurrent surgical and systemic intervention, may be necessary to optimize treatment success.