Aim: The aim of the present study was to present a case of maxillary aspergillosis with unusual clinical presentation and imaging findings. Background: The range of lesions and conditions that affect the maxillary sinus is wide and vast. This necessitates a dynamic multidisciplinary approach for proper diagnosis and effective management. Case description: We present an unusual case of maxillary aspergillosis that mimicked apical periodontitis and was most consistent with lymphoma from the diagnostic imaging perspective. Histopathology, however, established the correct diagnosis; appropriate management commenced promptly. Conclusion: It is imperative that maxillary aspergillosis be diagnosed and managed properly and promptly to improve prognosis. Advanced imaging is required, but it plays an adjunct role to histopathology. Long-term follow-up is also necessary to ensure complete resolution of the infection. Clinical significance: Correlating clinical and imaging findings is a crucial step and any inconsistencies should be resolved promptly so as not to delay adequate management. Histopathology often serves to dispute any inconsistencies and allows the establishment of a proper diagnosis.
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