Introduction: Paget's disease of the breast is a rare condition which is usually associated with underlying ductal in situ or invasive carcinoma. The incidence of tumor recurrence after mastectomy with preservation of the nipple-areola complex (NAC) is still unclear. Case Report: A 66-year-old woman diagnosed with a high-grade ductal carcinoma in situ underwent a nipple-areola sparing mastectomy, breast reconstruction with submuscular prosthesis, and adjuvant tamoxifen treatment. Eight years following breast surgery, the patient presented with an eczematoid lesion in the NAC. Histopathological exam was consistent with Paget's disease of the breast. Considerations: Only 14 cases of Paget's disease after nipple-areola sparing mastectomy have been published in the literature. The mean patient age reported was 48.8 years and disease occurred after a mean period of 44.6 months.

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