Acquired torticollis is not a separate disease rather a warning sign of an underlying disorder. The etiology of torticollis in children is quite wide. Hematogenous osteomyelitis of the first cervical vertebra (the atlas) is an extremely rare and potentially dangerous pathology. The aim is to acquaint physicians of different specialties with an extremely rare condition – acute hematogenous osteomyelitis of the atlas. Case Report. A case of osteomyelitis of the first cervical vertebra in a 7-year-old boy is presented. An important sign of the disease is torticollis. Conclusions. Hematogenous osteomyelitis of the atlas is an extremely rare and potentially dangerous pathology. Regardless of its incidence, it must be considered when examining a child with acquired wryneck. All the ENT specialists must possess the knowledge of this disease, as they are most likely the first doctors to see such a child. The research was carried out in accordance with the principles of the Helsinki Declaration. The informed consent of the patient was obtained for conducting the studies. No conflict of interest was declared by the authors. Key words: torticollis, osteomyelitis of the atlas, C1 osteomyelitis, vertebral osteomyelitis, children.