Squamous cell carcinoma (SCC) of the renal pelvis is an aggressive tumor with insidious onset of unspecific symptoms and advanced stages at diagnosis. It is a rare entity, accounting for 0.5-8% of renal tumors. In this paper, we describe the case of a patient with a history of recurrent nephrolithiasis that presented with an aggressive form of SCC of the renal pelvis with rapid relapse after resection.