Cystic hypersecretory carcinoma of the breast is very rare, and has a special histological morphology and immunophenotype. It was understood that it cannot be misdiagnosed as invasive carcinoma of the breast or other benign lesions. This paper reports a case of this tumor treated in our hospital on Apr. 2, 2011. The clinical data, ultrasonic manifestations, histomorphology and immunophenotype were analyzed retrospectively. This study aims to investigate the clinicopathological, immunohistochemistry, diagnosis and prognosis of cystic hypersecretory carcinoma of the breast, so as to improve clinicians’further understanding of it.