A 10-year retrospective analysis of invasive mold infections in hospitalized patients was performed to characterize the epidemiology and clinical features of invasive fungal sinusitis. Seventeen cases of invasive mold sinusitis were identified. Eleven cases were caused by Aspergillus flavus, three were caused by unspecified species, and one each was caused by Aspergillus fumigatus, Rhizopus species, and Alternaria species, respectively. Fifteen patients had hematologic malignancies, and two had end-stage liver disease. The most common presenting symptom was periorbital swelling (seven patients). Sinusitis was diagnosed a median of 19 days after admission. Eight patients (47%) survived; six of these patients were treated with both amphotericin B and surgery. Postmortem examination of six patients showed evidence of disseminated disease; the brain was the most common extrapulmonary site (four patients). To our knowledge, this is the largest currently reported series on invasive mold sinusitis; our report extends the information on invasive mold sinusitis and shows that aggressive therapeutic and surgical interventions are needed to prevent rapid progression of disease in immunocompromised patients.