LAUSR

BACKGROUND Cranioplasty is a well-established surgical operation that is used worldwide for patients with skull defects following decompressive craniectomy (DC). However, in some cases, potentially fatal complications may occur, such as malignant cerebral swelling after uneventful cranioplasty. CASE DESCRIPTION We present a rare case of massive malignant ipsilateral cerebral swelling following uneventful titanium mesh cranioplasty due to rare ipsilateral intracranial vasculopathy confirmed by magnetic resonance angiography (MRA) and magnetic resonance venography (MRV). Fortunately, we performed titanium mesh explantation and extended DC in time, and the patient survived. Malignant cerebral swelling after uneventful cranioplasty is an unpredictable but fatal complication. Most reported cases have had an unfavorable prognosis. To the best of our knowledge, the mechanism was first confirmed by MRA and MRV, which demonstrated that the cerebral swelling was due to unilateral intracranial vasculopathy, including a rare ipsilateral intracranial internal carotid artery occlusion, as well as extremely thin lateral and sigmoid sinuses. CONCLUSIONS Our case demonstrates for the first time that ipsilateral intracranial vasculopathy is a risk factor for malignant cerebral swelling after cranioplasty. Patients with traumatic brain injury with suspected intracranial vasculopathy should undergo a comprehensive vascular evaluation before cranioplasty to help prevent malignant cerebral swelling.