Abstract
BACKGROUND: Osteogenesis imperfecta (OI) is a connective tissue disorder characterized by fragile bones and vascular fragility, increasing the risk of vessel dissection and potentially complicating endovascular intervention. The authors present the first case of cranial bypass in a patient with OI. OBSERVATIONS: A 38-year-old male with OI type I presented with a symptomatic left internal carotid artery (ICA) occlusive dissection managed with endovascular revascularization and stenting. Follow-up surveillance imaging identified an incidental right ICA dissection, also treated with stenting. Four years later, the patient experienced new right hemispheric symptoms. He was found to have progressive right ICA dissection on best medical management. Following an unsuccessful restenting attempt, he underwent a successful double-barrel superficial temporal artery-to-middle cerebral artery (STA-MCA) bypass to restore cerebral perfusion with no perioperative complications. Six-month follow-up DSA confirmed a patent bypass with robust flow, and the patient remained asymptomatic 1 year postoperatively. LESSONS: STA-MCA bypass can serve as a viable and effective revascularization option in patients with OI, whose disease predisposes them to vascular dissection. In these high-risk patients, cranial bypass is a safe method for effective flow augmentation to hypoperfused brain regions when endovascular interventions fail. https://thejns.org/doi/10.3171/CASE25378.