Abstract： Objective To assess the clinical features of transperineal prostate biopsy in patients age ≤50 years.Methods The clinical data of 124 patients ≤ 50 years old were retrieved retrospectively in Peking Union Medical College Hospital between January 2005 and September 2014.The age of patients were 14 to 50 years (mean age 43.6 years),and their prostatic specific antigen(PSA) levels were fluctuated in a range of 0.01 to 579.00 μg/L (mean 15.5 μg/L).Twenty patients were abnormal in digital rectal examination (DRE).All the patients were underwent transperineal prostate biopsy using an 11-region template.Results Prostate cancer was detected in 14 of 124 patients (11.3％).The prostate cancer detection rates in groups with PSA 0-4.0,＞4.0-10.0,＞ 10.0-20.0,＞20.0-50.0,and ＞50.0 μg/L were 0,6.2％ (4/65),13.3％ (4/30),1/5,and 5/5,respectively.Non-adenocarcinoma prostate malignancy (NAPM) was detected in 7 of 124 patients (5.6％),and their PSA levels were fluctuated in a range of 0 to 4.0 μg/L Four patients were abnormal in DRE and 5 patients were abnormal in radiological examination.Conclusion The positive rate of transperineal prostate biopsy in patients age≤50 years is low,and rigorous screening before prostate biopsy is necessary.The men with DRE or radiological abnormalities but normal PSA should be wary of NAPM.