Introduction: Osteogenesis imperfecta (OI) is a genetic disease that primarily affects bone formation and metabolism. Craniofacial malformations belong to the broad spectrum of the OI phenotype. The introduction of bisphosphonate medications was intended to counteract the significant brittleness of the bones of OI patients. In connection with the application of bisphosphonates, drug-associated osteonecrosis of the jaw has become known as an undesirable effect of the therapeutically intended reduction of osteoclast activity. Originally, the pharmacological inhibition of bone loss was mainly used in oncological therapy. For some time now, osteoporosis has also been treated with substances that inhibit bone resorption. In OI, malposition of the jaws is relatively common, in particular retrognathia of the maxilla and progeny of the mandible. The literature discloses complications of orthognathic surgery in OI patients. Previous literature reviews suggest that bisphosphonate medication has no significant impact on the performance of and healing after oral surgery in OI patients. Material and methods: An essential prerequisite of a therapy adapted to the patient's condition is the knowledge of the patient's medical history. This case report describes the orthodontic-surgical treatment of an OI patient and the treatment experience derived in dealing with the condition. The unusual circumstance of this case is that the patient had concealed both his underlying disease and his medication during the current treatment period. In addition, the relevant literature is evaluated for combining the keywords OI, orthodontic therapy, bisphosphonates, and orthognathic surgery. Results: Based on the literature and our own experience, it is concluded that orthodontic treatment with bisphosphonate medication can also be carried out in OI patients. However, considerably greater forces and longer time intervals should be scheduled for each treatment. Orthognathic surgery with bisphosphonate medication turned out to be uncomplicated in our own case, considering the underlying disease in the planning of surgical procedures. However, there have been very few reports of OI patients in whom orthodontic-surgical treatment of jaw malformation has been performed with bisphosphonate medication. Conclusion: Taking into account the reported experience of severe complications of orthognathic surgery, the multiple documented adjustments to the treatment strategy of OI patients should be taken seriously. The basic condition of therapy adapted to the disease is that the patient informs the practitioner in an appropriate manner about his or her state of health.