Background: Granulomatous lobular mastitis (GLM) is a rare chronic inflammatory condition of the breast. The purpose of this study was to describe antituberculous treatment of GLM and the long-term follow-up outcome. Methods: This retrospective study included 22 patients who had been histopathologically diagnosed with GLM at the Second Hospital of Shandong University from January 2011 to March 2015. Clinical characteristics, ultrasonography and mammography findings, laboratory tests, treatment regimens, follow-up information, and recurrences were recorded. Results: All patients were female with a median age of 29 (range 23-44) years. The most common symptom was a breast mass with or without pain. Large irregular hypoechoic masses could be found in the breast ultrasounds of 13 patients. All patients received triple antituberculous therapy. During a median follow-up period of 40 months, 3 patients were lost to follow-up; of the remaining 19 patients, 18 achieved clinical complete remission and no recurrences were observed. Conclusion: GLM is an unusual benign breast condition that mimics breast carcinoma in its clinical and imaging presentation. Antituberculous therapy seems to be an effective alternative option in the treatment of GLM.