The study describes isolation of Neoscytalidium dimidiatum from a case of eosinophilic fungal rhinosinusitis (EFRS). The isolate was identified by routine mycological methods and confirmed by DNA sequencing of the internal transcribed spacer (ITS) region of rDNA. To our knowledge, this is the first report of its kind from Iran.The study describes isolation of Neoscytalidium dimidiatum from a case of eosinophilic fungal rhinosinusitis (EFRS). The isolate was identified by routine mycological methods and confirmed by DNA sequencing of the internal transcribed spacer (ITS) region of rDNA. To our knowledge, this is the first report of its kind from Iran.