Medical therapy of maxillary sinus inflammatory myofibroblastic tumors




Abstract


Inflammatory myofibroblastic tumor (IMT) in the maxillary sinus is a diagnostic challenge. As IMT has various names, it has various findings in magnetic resonance image. Although destructive pattern in computed tomography and hypermetabolism in PET CT suggest malignancy, it is debatable whether it is a tumor or inflammatory lesion. Treatment of IMT usually includes surgery. However, IMT can be dealt with medical treatment according to histologic type and localization. We report a rare case of IMT in the maxillary sinus which is controlled by medical therapy.



Introduction


Inflammatory myofibroblastic tumor (IMT) is also referred to as inflammatory pseudotumor, plasma cell granuloma, benign myofibroblastoma, xanthogranuloma, fibrous histiocytoma, and atypical fibromyxoid tumor due to its variable cellular components. This disease is a rare tumorous condition in that it has characteristics of invasion, local recurrence and chromosomal abnormality, although whether it is a tumor or reactive lesion is debatable .


Histologic diversity complicates diagnosis. Although there is no standard treatment, this tumor is usually treated by surgery . However, IMT can be treated by medication according to the histologic type.


We present a rare case of IMT in the maxillary sinus that was controlled by medical therapy.





Case report


A 60-year-old woman presented to our hospital with left nasal obstruction, anosmia, and frequent epistaxis. Physical examination showed left cheek tenderness and swelling, and rhinoscopy revealed an irregularly shaped mass in the left nasal cavity ( Fig. 1 ). She had no remarkable medical history and had no ocular symptoms. Computed tomography (CT) revealed that the soft tissue mass was opacified in the left maxillary sinus and bony destruction was noted ( Fig. 2 A ). Magnetic resonance imaging (MRI) revealed an isosignal in the T1 weighted image and mild hyperintensity in T2 weighted image. The mass invaded the pterygoid muscles and pterygoid plate posteriorly ( Fig. 2 B) Positron emission tomography (PET) CT revealed the hypermetabolic mass in the left maxillary sinus (peak SUV = 12.10) and invasion to the pterygoid muscle ( Fig. 2 C).




Fig. 1


Endoscopic view of nasal cavity. Huge irregular mass was occupied from the left nasal cavity.



Fig. 2


A. Axial computed tomogram (left); coronal computed tomogram (right). The images show a mass involving left maxillary sinus, extending into left orbit and destructing anterior wall, posterior wall and superior wall. B. MRI showed that isosignal in the T1 weighted image (left) and mild high signal in T2 weighted image (right) were noted. The mass was invaded the pterygoid muscles and pterygoid plate posteriorly. C. PET CT revealed that hypermetabolic mass was noted in the left maxillary sinus (peak SUV = 12.10) and invaded to the pterygoid muscle.


We suspected a malignant tumor in the left maxillary sinus and performed a Caldwell Luc biopsy by local anesthesia. The biopsy result showed atypical cells and chronic inflammation. To obtain the correct diagnosis, we did same biopsy under general anesthesia. The final biopsy was positive for smooth muscle actin and epithelial membrane antigen, and negative for S-100 protein and anaplastic lymphoma kinase. The results indicated an inflammatory myofibroblastic tumor ( Fig. 3 ).




Fig. 3


Left: Prominent lymphocytes and plasma cells infiltrate among the bland spindle cells (H&E × 400). Right: Immunostain demonstrates that the spindle cells express smooth muscle actin.


The patient received medical treatment consisting of oral prednisolone (1 mg/kg) for 7 months and oral methotrexate (15 mg/wk) for 2 years. A 3-year follow-up was uneventful and no lesion of nasal cavity and maxillary sinus was detected ( Fig. 4 ).


Aug 23, 2017 | Posted by in OTOLARYNGOLOGY | Comments Off on Medical therapy of maxillary sinus inflammatory myofibroblastic tumors

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