Testicular microlithiasis is an uncommon condition in which multiple calcifications develop in the seminiferous tubules. Sonography demonstrates a typical pattern of diffuse, hyperechoic specks. This condition has been associated with premalignant and malignant changes of the testicle. We present a case of intratubular germ cell neoplasia and a microscopic focus of seminoma found in a patient with isolated testicular microlithiasis and no focal mass. Physicians should be more aware of testicular microlithiasis, and heightened surveillance is recommended to identify progression to tumor formation. Copyright 1996 by Elsevier Science inc.