- Objective Acute invasive fungal sinusitis (AIFS) appears mainly in immunocompromized patients and may be caused by various pathogens. We describe a teenager with invasive sinonasal Scopulariopsis brevicaulis and review all the reports on this rare pathogen. Methods A literature search on Scopulariopsis sinonasal invasive infections was performed and clinical data including age, gender, co-morbidities, treatment and prognosis was collected on all the patients. Results A 17 years old boy with acute myelocytic leukemia and Scopulariopsis brevicaulis sinonasal infection was successfully treated at our department with a combination of extensive surgical debridement and antifungal antibiotics. We found six articled describing six patients with AIFS due to Scopulariopsis species. Four patients were adults and two were children, 3 males and 3 females. Two had an infection with Scopulariopsis acremoium , one with Scopulariopsis candida and for 3 patients no data was given on the specific Scopulariopsis species. All the patients except one were immunocompromized. One patient was treated with antifungal drugs, 2 with surgery and 4 patients received antifungals and were operated. One patient died due to the fungal infection and two patients died due to other causes. Conclusions Scopulariopsis AIFS is a life threatening disease affecting mainly immunocompromized patients, both children and adults. No clear treatment regimen has been established yet. We describe the first case of a teenager with Scopulariopsis brevicaulis sinonasal infection treated successfully with a combination of wide local excision and antifungal therapy.