Introduction: Adenoid cystic carcinoma (ACC) is a rare, slow-growing malignant tumor, mostly occurs in the minor salivary glands. Moreover, ACC arising in the sphenoid sinus is a rare entity. Case: Herein, we report a case of ACC in a 69-year-old man who presented with a headache and diplopia. Both computed tomography (CT) and magnetic resonance imaging (MRI) demonstrated a mass lesion in left sphenoid sinus extending to the sellar area and clivus. After resection of the tumor, histopathological examination and immunohistochemical analysis reported "adenoid cystic carcinoma". Conclusion: We introduced a case of ACC that presented with a headache and diplopia. Although rare, ACC can arise from sphenoid sinus and should be considered as differential diagnosis of sphenoid lesions.
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