The brown tumor of hyperparathyroidism is a rare condition associated with excessive production of parathyroid hormone in response to decreased serum calcium levels associated with chronic kidney disease (CKD). A 28-year-old female patient with CKD undergoing hemodialysis presented an asymptomatic swelling in the buccal region of teeth 34 to 44 and mobility of them. Similar swellings were observed on the palate and alveolar ridge distal to tooth 17. On the panoramic radiograph, a radiolucent area not defined by radiopaque halo was observed in the anterior region of the mandible of approximately 2.5 cm in length and radiolucent area around tooth 46. Biochemical tests showed hyperphosphatemia, hypercalcemia, and hyperparathyroidism. An incisional biopsy was performed, and the fragments removed were submitted to histologic analysis, which showed multiple multinucleated giant cells in hemorrhagic exudate. The patient was referred to the medical service for treatment of hyperparathyroidism secondary to CKD. The brown tumor of hyperparathyroidism is a rare condition associated with excessive production of parathyroid hormone in response to decreased serum calcium levels associated with chronic kidney disease (CKD). A 28-year-old female patient with CKD undergoing hemodialysis presented an asymptomatic swelling in the buccal region of teeth 34 to 44 and mobility of them. Similar swellings were observed on the palate and alveolar ridge distal to tooth 17. On the panoramic radiograph, a radiolucent area not defined by radiopaque halo was observed in the anterior region of the mandible of approximately 2.5 cm in length and radiolucent area around tooth 46. Biochemical tests showed hyperphosphatemia, hypercalcemia, and hyperparathyroidism. An incisional biopsy was performed, and the fragments removed were submitted to histologic analysis, which showed multiple multinucleated giant cells in hemorrhagic exudate. The patient was referred to the medical service for treatment of hyperparathyroidism secondary to CKD.