Spindle cell carcinoma is a rare malignancy of the head and neck region. It is a biphasic neoplasm consisting of epithelial and mesenchymal components and accounts for less than 1% of all tumours of orofacial region. It is a unique variant of squamous cell carcinoma which is very aggressive in nature and associated with relatively poor prognosis. The most common site for occurrence is upper aerodigestive tract, esophagus, salivary glands, thyroid, thymus, lung, breast, gastrointestinal tract, hepatobiliary system, genitourinary tract and uterus. Involvement of maxillary sinus by spindle cell carcinoma is a rare occurrence and follows aggressive course with a high mortality rate. We report a rare case of spindle cell carcinoma of maxilla with immunohistochemical studies. We present a case of spindle cell carcinoma involving right maxillary antrum.