This case report describes a 21-year-old female patient with a complex medical condition of end-stage chronic renal failure and secondary hyperparathyroidism presenting with a history of gradual enlargement of the facial bones over a period of one year. The facial enlargement primarily involves the maxilla causing a bizarre facial and dental deformity. Based on the clinical, radiographic, and laboratory investigations the facial deformity was confirmed as a rare manifestation of renal osteodystrophy presenting as maxillary hyperplasia and hyperostosis cranialis.