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To describe the oral and maxillofacial manifestations of patients diagnosed with chronic kidney disease–mineral and bone disorders.Over a 13-year period, clinicopathologic data of patients diagnosed with CKD-MBD who had oral and maxillofacial alterations were retrieved from the files of 4 Brazilian institutions. Data included clinical, radiographic, microscopic, and biochemical findings; treatment employed; and follow-up status.Twenty-one cases were identified, with 13 patients diagnosed as brown tumor of hyperparathyroidism (BTH) and 8 as osteitis fibrosa/renal osteodystrophy (OF/RO) (4 of them clinically consistent with Sagliker syndrome). The mean age was 32.7 years, and the mandible was the most affected site (42.8%). OF/RO had an ill-defined “ground glass” radiographic appearance, and BTH produced well-defined radiolucent images. Biochemically the following mean values were obtained: parathormone 1511.07 pg/mL, calcium 9.25 mg/dL, phosphorus 5.19 mg/dL, alkaline phosphatase 941.55 U/L, urea 125.42 mg/dL, and creatinine 7.14 mg/dL. Treatment comprised vitamin D and calcium intake, parathyroidectomy, hemodialysis, renal transplantation, and local surgery. During follow-up, 5 patients with BTH were free of lesions, whereas 2 affected by OF/RO/Sagliker syndrome died.Oral and maxillofacial manifestations of BTH and OF/RO are uncommon, but they can be associated with an important life-threatening scenario.