Phaeoacremonium parasiticum is an unusual cause of human fungal infection that was first reported in 1974. Since the initial description, only a small number of cases have been reported in the world literature. We report on the clinical and imaging findings of Phaeoacremonium parasiticum myositis in a renal transplant recipient. To our understanding, this is a first case report of Phaeoacremonium parasiticum myositis that includes imaging findings.