Orthodontic therapy in patients with osteopetrosis (OP) of the jaws has typically been contraindicated owing to the presence of poorly perfused and extremely compact bone, and the potential risk for infection and osteomyelitis. As such, completed orthodontic cases in association with OP have rarely been published. A patient aged 14 years 6 months, with no known diagnosis of OP, sought orthodontic assessment for anterior crowding. The patient exhibited a straight facial profile and increased mandibular facial height, competent lips, shallow mentolabial sulcus with mild mentalis strain, flat/reverse smile arc and wide buccal corridors on smiling. The patient had a Class I incisor relationship on Class I skeletal bases with bilateral Class I molars and Class II canine relationships. This was complicated by a crossbite involving the lateral incisors and a Bolton discrepancy due to small maxillary lateral incisors. A radiologic assessment revealed polyostotic OP of the oromaxillofacial complex. Treatment consisted of maxillary and mandibular fixed orthodontic therapy, bite turbos and elastics to level and align the dentition. Extractions of permanent teeth were not needed. At the conclusion of treatment, there was a slight left Class II malocclusion, with incomplete intercuspation on the left side due to tooth size discrepancy, possibly attributed to inadequate elastics compliance and the presence of osteopetrotic bone. The treatment was completed in 3 years, 1 year longer than anticipated. This report represents the second published account of a patient with OP successfully managed with comprehensive orthodontic care and without osseous complications. Obtaining cephalometric measurements on OP-affected patients may be imprecise owing to the presence of extremely dense bone and difficulty to identify bony landmarks. To reduce osteopetrotic sequelae, attending clinicians should consider reduced exertional orthodontic forces and closely monitor patients for adverse alveolar events.