BACKGROUND Concomitant allergic fungal rhinosinusitis(AFRS) and allergic bronchopulmonary aspergillosis(ABPA) are extremely rare, with no more than20 cases reported in the English literature.CASE SUMMARY A 52-year-old female patient complained of right-sided nasal obstruction,rhinorrhea, sneezing, epistaxis, and hyposmia for a period of around 5 mo. Nasal examination detected paleness and edema of the nasal mucous membrane and a polyp in the right middle meatus. A computed tomography(CT) scan of the sinuses revealed a ground-glass opacity filling the right maxillary and ethmoid sinuses, along with bone absorption in the medial wall of the right maxillary sinus. Magnetic resonance images were obtained with T1-weighted, T2-weighted,and gadolinium-enhanced T1-weighted sequences. A well-defined mass, located in the right maxillary and ethmoid sinuses and displaying obvious hypointense features, was observed on both T1-and T2-weighted images, with peripheral enhancement on gadolinium-enhanced T1-weighted images. The patient also has a 20-year history of cough and dyspnea. Chest CT revealed columned and cystiform bronchiectasis in the bilateral bronchus, surrounded by a large number of spotted and funicular high-density lesions. The level of serum total Ig E was >5000 k U/L. Serum Ig E levels related to house dust and aspergillus showed a positive result, with the values being 3.5 k U/L and 1.2 k U/L. We performed functional endoscopic sinus surgery under local anesthesia. After surgery, topical glucocorticoids and saline irrigation were applied in the nasal cavity until the present time. An oral glucocorticoid(methylprednisolone 16 mg/d) and antifungal agent(itraconazole 200 mg/d) were also used for a period of 4 wk.Montelukast was prescribed at 10 mg/d until the present time. An endoscopic examination showed that the patient was recovering well at 3 mo after surgery.CONCLUSIONSince different specialists treat ABPA and AFRS, their coexistence may be overlooked. AFRS accompanied by ABPA requires surgical therapy combined with medical control to improve the symptoms.