Background Phyllodes tumor is rare tumor of the breast, which encompasses both stromal and epithelial components. In these components, metaplastic changes can be observed occasionally. Case presentation We report the case of a 51-year-old woman nulligest menopaused who presented a huge mass, largely ulcerated in her right breast. The radiological examination revealed a large tumor with microcalicifications classified as Breast Imaging and Reporting Data System [BI-RADS] category 5. The patient undergone right mastectomy and the histological analysis revealed benign phyllodes tumor with cystic squamous metaplasia. Conclusions It is essential to be aware of this uncommon event in order to avoid misdiagnosis.