Transplant renal artery stenosis (TRAS) is a potentially treatable cause of allograft dysfunction, hypertension and graft loss. The mainstay of treatment includes angioplasty and endovascular stenting, although observation and surgery are at times indicated. We present an unusual case of TRAS secondary to mechanical compression from a patient's enlarged native polycystic kidneys. This was treated with bilateral native nephrectomy and evidence of TRAS improved both clinically and radiographically. Recognition and treatment are important in preventing irreversible complications of TRAS.