The authors present a new case of intratesticular varicocele(ITV). The ultrasound findings on gray-scale and color Doppler imaging as well as the pertinent clinical manifestations are discussed. A review of the literature is also presented. According to our knowledge only 34 cases of ITV have been reported in the literature and the clinical significance of this lesion is not yet well established. This is the first reported case of infertility caused by a solely ITV, showing that the clinical implications of this entity may be more significant than previously thought.