A 34 years old male patient with chronic renal failure and history of total parathyroidectomy with parathyroid autotransplantation evolved with persistence of elevated PTH, bone pain. Cervical ultrasonography and parathyroid scintigraphy with sestaMIBI revealed changes in the topography of the left submandibular gland. He underwent cervical exploration showing supranumerary ectopic parathyroid, found in the left carotid sheath at I/IIA levels, with histopathological confirmation by frozen section biopsy. On the first postoperative day, serum PTH decreased by more than 95%, thus, confirming operative success. The symptoms improved along the follow up.A 34 years old male patient with chronic renal failure and history of total parathyroidectomy with parathyroid autotransplantation evolved with persistence of elevated PTH, bone pain. Cervical ultrasonography and parathyroid scintigraphy with sestaMIBI revealed changes in the topography of the left submandibular gland. He underwent cervical exploration showing supranumerary ectopic parathyroid, found in the left carotid sheath at I/IIA levels, with histopathological confirmation by frozen section biopsy. On the first postoperative day, serum PTH decreased by more than 95%, thus, confirming operative success. The symptoms improved along the follow up.