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Abstract Pilomatrix carcinoma is a very rare malignant tumor. The present article raises awareness of the clinical features, diagnosis, and treatment of this malignancy. The clinical data of a case of pilomatrix carcinoma of the left temporal region treated at the First Affiliated Hospital of Nan Chang University, Nan Chang, China, in April 2017 were analyzed retrospectively, and the related literature was reviewed. The patient underwent a wide excision of the mass, and microscopic examination revealed that the tumor showed the typical features of pilomatrix carcinoma. Pilomatrix carcinoma is a very rare tumor with approximately 130 cases reported in the English literature. An optimal treatment regimen has not been established; however, wide surgical excision is recommended to avoid recurrence when the staging shows no metastasis, and adjuvant radiotherapy might be beneficial for local tumor control. Pilomatrix carcinoma is considered to be a low‐grade malignant tumor and shows an aggressive growth pattern. A wide surgical excision and radiotherapy are the main modalities of treatment.