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Background: Craniectomies represent a lifesaving neurosurgical procedure for many severe neurological conditions, such as traumatic brain injury. Syndrome of trephined (SoT) is an important complication of decompressive craniectomy, and cranial reconstruction is the definitive treatment. However, many patients cannot undergo surgical intervention because of neurological status, healing of the primary surgical wound, or the presence of concurrent infection, which may prevent cranioplasty., Objective: To offer a customized external cranioplasty option for managing skull deformities for patients who could not undergo surgical intervention for definitive cranioplasty., Methods: We describe the design and clinical application of an external cranioplasty for a patient with a medical history of intractable epilepsy, for which she underwent multiple right cerebral resections with a large resultant skull defect and SoT., Results: The patient had resolution of symptoms and restoration of a symmetrical skull contour with no complication at 17 months., Conclusion: Customized external cranioplasty can improve symptoms associated with SoT for patients who cannot undergo a definitive cranioplasty. In addition, inset monitoring options, such as electroencephalography or telemetric intracranial pressure sensors, could be incorporated in the future for comprehensive monitoring of the patient's neurological condition., (Copyright © Congress of Neurological Surgeons 2023. All rights reserved.)