Testicular microlithiasis is a relatively uncommon condition in children. It is characterized by the presence of microcalcifications within the testicular parenchyma. Although it is a benign finding, underlying diseases and other conditions must be ruled out because testicular microlithiasis has been found in association with both benign and malignant lesions in the testes and other tissues. We present two brothers with testicular microlithiasis, and highlight the prevalence, natural history, associated malignant conditions and follow-up recommendations of children diagnosed with testicular microlithiasis.