Sebaceous carcinoma (SC) is an uncommon but aggressive malignancy and has a specific anatomic preference for the ocular region, especially the eyelids. However, periocular SC originated from the eyebrow is rare, which may cause poorer outcomes due to a greater likelihood of orbital invasion and excessive tumor volume. In the present case, we exhibited a 68-year-old male presenting with a large solid mass in his right eyebrow region developing in ten months. Based on the patient's history, clinical conditions, orbital computed tomography (CT) and magnetic resonance imaging (MRI) scan results, a malignant tumor was suspected preliminarily. Excisional biopsy was performed, and the histopathologic examination and immunohistochemistry (IHC) staining of the tumor revealed SC. The patient declined the enlarged surgery recommended next and ended up with death caused by the distant metastasis of SC. The case highlighted the fact that despite its rarity, SC should be considered as a differential diagnosis of tumors located in the eyebrow region and histopathologic evaluation must be performed to reach a definite diagnosis. Ophthalmologists are supposed to have a comprehensive understanding of the clinicopathological characteristics of this disease and help patients accept the appropriate treatments promptly via properly and adequate communication if necessary.