Background: Transduodenal ampullectomy (TDA) is a less invasive procedure than pancreaticoduodenectomy (PD). However, the outcomes of TDA and PD have been compared rarely in early ampullary cancer. Methods: From September 1994 to June 2013, the patients who underwent curative surgery for Tis or T1 ampulla of Vater neoplasm were identified. The patients were divided into two groups according to the types of surgery; TDA group and PD group. The patient characteristics and survival outcomes were retrospectively investigated between the two groups. Results: Total 137 patients were included in this study. The 18 patients underwent TDA and 119 patients underwent PD for Tis or T1 ampullary cancer. There was no lymph node metastasis in the patients with Tis tumor although 10 of 104 patients had lymph node metastasis in T1 cancer. After a median follow-up of 50 months (range, 6-148), there were no recurrence after TDA for Tis tumor. However, the TDA was associated with higher local recurrence rate than PD in the patients with T1 ampullary cancer on Kaplan-Meier survival analysis (p = 0.007). Conclusion: The TDA is feasible treatment for Tis ampulla of Vater neoplasm. However, TDA is unsuitable for the treatment of T1 ampulla of Vater cancer.