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Abstract Background Inflammatory myofibroblastic tumor (IMT) is a mesenchymal tumor mainly found in lung or retroperitoneum and rarely affects head and neck region. Extensive English literature search reveals that less than fifty cases of head and neck IMT have been reported so far, maxillary sinus being fewer. Case presentation We present a case of IMT involving maxillary sinus in a 48-year-old gentleman who attained complete clinico-radiologic response after treatment with radiotherapy (RT) and concurrent oral prednisolone. Conclusions This is the first report where such magnificent response was attained in primary setting treated with RT and steroids as opposed to surgery which used to be considered as standard of care till now.