Neoscytalidium dimidiatum is a plant pathogen, but can also cause onychomycosis. We compared clinical and epidemiological data of cases of onychomycosis caused by N. dimidiatum and Trichophyton rubrum. We also evaluated the in vitro antifungal susceptibility of N. dimidiatum clinical isolates. It was not possible to establish any statistical differences between groups, except the place of residence and the number of affected nails. The results suggest that onychomycosis caused by N. dimidiatum is clinically similar to that caused by T. rubrum; besides, N. dimidiatum has been shown to have low sensitivity to itraconazole, but high to terbinafine. Lay Summary Cases of onychomycosis caused by Neoscytalidium dimidiatum were studied and compared to cases of onychomycosis caused by T. rubrum. The individuals affected were adults, and the clinical characteristics were not different between groups; accordingly, mycological diagnosis is mandatory.