Simplicilliumspecies are widely distributed with a broad spectrum of hosts and substrates. Generally, these species are entomopathogenic or mycoparasitic. Notably, some isolates ofSimplicillium lanosoniveumandSimplicillium obclavatumwere obtained from human tissues. In this study, two fungi were isolated from the annular itchy patch of infected skin of a 46-year-old man with diabetes mellitus. Based on a combination of morphological characteristics and phylogenetic analysis, a novel species,Simplicillium sinense, was introduced herein. It morphologically differs from the remainingSimplicilliumin the size of phialides and conidia. Additionally, it grows slowly on YPD at 37°C. Antimicrobial susceptibility testing presented that this fungus is resistant to most azole antifungals. Therefore, the diagnosis of tinea faciei was made, and after 2 weeks of being treated with oral terbinafine (250 mg, once a day) and topical terbinafine cream for 1 month, the rash was mainly resolved and no recurrence happened after 6 months of follow-up. Herein,Simplicillium sinensewas introduced as a new fungal taxon. Meanwhile, a case of superficial infection caused byS. sinensewas reported. So far, it is the thirdSimplicilliumspecies obtained from human tissue. Meanwhile, terbinafine is recommended as the first-line antifungal treatment againstSimplicilliuminfection.