A case of allergic fungal sinusitis (AFS) caused by Exserohilum rostratum, proven by culture and histopathology of the biopsy material, is described. The identity of the isolate was confirmed by sequencing of ITS region of rDNA. To the best of our knowledge, this is the first report of allergic E. rostratum sinusitis from Kuwait and Arabian Peninsula. Ten previously described cases of AFS have been reviewed. The report highlights the emerging importance of E. rostratum as a human pathogen in this region and role of molecular methods in its accurate identification.