The malignant proliferating trichilemmal tumor (MPTT) is an exceptional adnexal neoplasm of the skin, primarily observed in the scalp of elderly women, often precipitated by local trauma or chronic inflammation. Notably, MPTT exhibits a propensity for local aggressiveness, frequently leading to diagnostic challenges reminiscent of squamous cell carcinoma. The primary treatment modality entails surgical excision with adequate margins, yielding a relatively low local recurrence rate. Herein, we present the case of a 50-year-old woman diagnosed with MPTT of the scalp several years subsequent to undergoing chemotherapy for breast cancer, initially misdiagnosed as squamous cell carcinoma upon biopsy. This case marks only the third reported instance associating MPTT with chemotherapy following breast cancer treatment. We aim to draw attention to this particularly aggressive tumor and its potential origination from benign trichilemmal cysts due to chemotherapy.