Decompressive craniectomy is an established procedure to lower intracranial pressure and can save patients' lives. However, this procedure is associated with delayed cognitive decline and cerebral hemodynamics complications. Studies show the benefits of cranioplasty beyond cosmetic aspects, including brain protection, and functional and cerebrovascular aspects, but a detailed description of the concrete changes following this procedure are lacking. In this paper, the authors report a patient with trephine syndrome who underwent cranioplasty; comprehensive cognitive and cerebral hemodynamic evaluations were performed prior to and following the cranioplasty. The discussion was based on a critical literature review.