Carcinoma in situ of the male breast is rare, especially when associated with bilateral gynaecomastia and during puberty. Only two cases in adults and one during puberty have been described in the literature. We present the case of a 15-year-old boy whom we have been observing for about 2 years for puberal bilateral gynaecomastia, treated with bilateral exeresis of the mammary gland and subsequently re-operated with total bilateral mastectomy because the tissue removed presented atypical features upon histological examination. The authors believe, after a careful review of relevant literature, that this is the second case of intraductal carcinoma in the presence of gynaecomastia described during adolescence.