Abstract We report a case of a 19-year-old man who presented with severe scrotal swelling. His medical history was notable for severe global edema at birth. Most areas of swelling had resolved by adolescence with exception of the scrotum and the left lower extremity. 99mTc-filtered sulfur colloid lymphoscintigraphy of the lower extremities demonstrated prominent dermal backflow into the superficial scrotum and thighs, which were confirmed on the SPECT/CT images, and correlated with findings on MR lymphangiogram. Lymphoscintigram and MR lymphangiogram may provide complementary information to aid diagnosis and management of primary scrotal lymphedema.