Introduction. Chronic granulomatous invasive fungal rhinosinusitis (CGIFRS) is an extremely rare form of invasive fungal sinusitis. The disease has a long-lasting and indolent course, so the invasion and destruction in the sinonasal and adjacent regions is progressing slowly. This disease has been reported primarily in Middle East, North Africa, India and Pakistan; however, it is very rare in western countries. It is primarily caused by Aspergillus flavus. Case Report. A 40-year-old man, who was repeatedly surgically treated for chronic rhinosinusitis with nasal polyps, was presented to our Otorhinolaryngology Department with a progressive, sudden right-sided proptosis. Contrast-enhanced paranasal sinus computed tomography (CT) showed almost complete soft tissue opacification of the sinonasal region, eroded bone structures and expansion into the right orbit. Erosion of the walls of the right frontal sinus was also seen but without intracranial propagation. Histopathological examination was necessary for the final diagnosis. The finding of granulomatous response along with fibrosis and strong inflammatory infiltrate was typical for chronic granulomatous invasive fungal sinusitis. The patient was successfully treated with a combination of surgery and postoperative medical therapy with voriconazole. To our knowledge, this is the first case of CGIFRS presented in Serbia. In addition, we reviewed the literature concerning this rare form of fungal sinusitis, especially for the Balkan region. Conclusion. Although we reported an extremely rare case of fungal sinusitis for the Balkan region, it is important to suspect on it in all cases where chronic inflammation of the paranasal sinuses does not respond to conventional treatment.
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