Introduction: Primary squamous cell carcinoma of the kidney is a rare tumor with a poor prognosis. It often occurs on a lithiasis kidney. Observation: We report a case of a 62-year-old patient admitted for left back pain associated with lower urinary tract symptomes with fever. Clinical examination revealed tenderness of the left lumbar fossa. Radiological examinations showed left ureterohydronephrosis, with the presence of a left kidney stone. A left radical nephrectomy was performed showing the presence of a neoplasm measuring 7cm with on histological examination a moderately differentiated and infiltrating squamous cell carcinoma. Conclusion: The diagnosis of squamous cell carcinoma of the kidney must be evoked in the first place in front of any renal mass with notion of kidney stones.