A 37-year-old woman underwent a total mastectomy and adjuvant chemotherapy for HER2-positive breast cancer (pT1N0M), and then recurred in the right lung followed by the pancreas. Lung lobectomy and pylorus-preserving pancreaticoduodenectomy were performed, and systemic chemotherapies including trastuzumab were sequentially administered. However, metastasis to the pancreatic tail was detected. She underwent image-guided radiation therapy, but this was not effective. Lapatinib plus capecitabine combination was administered as forth-line treatment and the metastatic lesion was disappeared. She is continuing this regimen with a complete response for 48 months until now.

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Intraductal tubulopapillary neoplasm (ITPN) of the pancreas has been recently reported. It is very rare, therefore clinical behavior and prognosis has not yet been characterized. We experienced a case of ITPN of the pancreas which presented with acute pancreatitis and treated with Whipple's operation. Histopathologic finding showed papillary hyperplasia with carcinomatous change. The tumor recurred after 47 month of operation, and she underwent total pancreatectomy. Pathologic finding revealed tubulopapillary growth with high grade dysplasia. Immunohistochemial staining was not performed, however gross and microscopic findings were compatible with ITPN of the pancreas. We report a case of ITPN of the pancreas.