Osteogenesis imperfecta (OI), is a heterogeneous group of inherited disorders of connective tissue characterized by bone fragility. Patients with this disease frequently suffer fractures, over 80% of the extremities due to the more intensive mechanical load. Fractures of the facial bones occur very infrequently. Several studies have proved that bisphosphonate therapy may be effective in reducing fracture risk in OI patients. We report here an unusual case of pathological mandibular fracture following simple molar extraction in an adult patient with OI type I and oral alendronic acid treatment. Extraction was atraumatical and without bone removal. Fracture was treated by internal fixation by plate osteosynthesis and additional mandibulomaxillary fixation (MMF). Bone healing after fracture treatment was completely normal and no clinical signs of osteoneocrosis were observed. To the best of our knowledge, this is an unusual complication even in this type of patients. Particular care is necessary is these patients during oral surgery, even when they have been treated and controlled.