Ostitis in the course of secondary hyperparathyroidism can lead to heavy atypical skeletal changes. In 4-year old girl on hemodialysis, with hyperparathyroidism (iPTH level > 1500 pg/ml), despite specific pharmacological treatment, very pronounced skull and maxillary bone malformations developed within two months, which were the cause of problems with breathing. We observed also growing bones malformations concerning arms, legs, vertebral column and pelvis. Severe infectious ostitis with severe uremic osteodystrophy was diagnosed. After 3 months of antibiotic treatment (clindamycin) subtotal parathyreidectomy was performed. Control iPTH level was 111 pg/ml. In next weeks skull skeletal changes partially resolved, which was confirmed on computer tomography, and patients general condition improved. She has no longer difficulties with breathing.