Purpose: With an increase in the population of immunocompromised patients, the incidence of maxillary sinus aspergillus infection has also escalated. Maxillary sinus aspergillosis is generally extended to the sinus antrum, base or thin orbital wall and ethmoid air cell region. We experienced a case of maxillary sinus aspergillosis which was extended directly to the soft tissue of the cheek. Methods: A 46-year-old man with acute myelogenous leukemia was consulted for the defect of the anterior wall of the maxillary sinus, and cheek. Radiologic and histologic findings were consistent with invasive maxillary sinus aspergillosis. The otolaryngology department performed debridement via endoscopic sinus surgery first. Coverage of the resulting defect in the anterior wall of the maxillary sinus and its inner layer was undergone by the plastic and reconstructive surgery department, using a pedicled superficial temporal fascia flap and a split thickness skin graft. The remaining skin defect of the cheek was covered with a local skin flap. Results: The patient went through an uneventful recovery. There was no recurrence during 6 months of follow-up. Conclusion: Maxillary sinus aspergillosis usually involves the orbit or the gingiva but in some cases it may directly invade soft tissues of the cheek. Such an atypical infection extending into the cheek may lead to a large soft tissue defect requiring coverage. Thus, any undiagnosed soft tissue defect involving the cheek or maxillofacial area, especially in immunocompromised patients, should be evaluated for aspergillosis. We present this rare case, with a review of the related literature.