Sagliker syndrome (SS), also known as an "uglifying human face syndrome," is one of the severe manifestations of chronic kidney disease in patients with uncontrolled secondary hyperparathyroidism. Patients with SS develop short stature, maxillary and mandibular bone overgrowth, nasal bone and cartilage destruction, widely spaced teeth with anterior positioning, soft tissue tumors in the oral cavity, auditory loss, and neurological and psychological features. SS can possibly be prevented with proper treatment of secondary hyperparathyroidism using disease-modifying medication and surgical therapies. We report a case of SS in a patient in the USA with end-stage renal disease achieving adequate hemodialysis.