Background <p>Cerebral venous air embolism is a rare but potentially fatal complication of central venous catheter insertion and removal. Although most venous air emboli remain clinically silent or cause only transient symptoms, they may exceptionally lead to extensive arterial cerebral ischemia, particularly in patients with impaired pulmonary filtration or pre-existing lung disease. We report a distinctive sequence of computed-tomography-demonstrated cortical venous air followed by extensive right middle and posterior cerebral artery infarction on magnetic resonance imaging despite a negative saline-contrast echocardiography for intracardiac shunt, occurring after elective internal jugular central venous catheter removal in a frail patient with idiopathic pulmonary fibrosis.</p> Case presentation <p>We report the case of a 68-year-old Lebanese, right-side dominant, cachectic man with multiple comorbidities, including coronary artery disease, heart failure with reduced ejection fraction, pulmonary fibrosis on long-term oxygen therapy, chronic kidney disease, and a history of nephrectomy for metastatic renal carcinoma. He was admitted for weight loss and hypotension, diagnosed with cardiogenic shock, and managed with vasopressor support in the intensive care unit. After stabilization and transfer to the ward, removal of a right internal jugular central venous catheter led to the sudden onset of acute paraplegia, altered consciousness, and left-sided neurological deficits. A non-contrast brain computed tomography scan revealed multiple cortical venous air emboli, and magnetic resonance imaging confirmed extensive ischemic lesions in the right-middle and posterior cerebral artery territories. Despite immediate supportive measures and antiseizure treatment, the patient developed refractory status epilepticus. Given his advanced comorbidities, extensive cerebral injury, and poor pre-morbid status, a shared decision was made with the family to limit further aggressive treatment, and the patient died a few hours later.</p> Conclusion <p>This case suggests that venous cortical air embolism can be a hidden precursor of extensive arterial ischemia even without an intracardiac right-to-left shunt, particularly when pulmonary filtration reserve is limited. Clinicians should maintain a high index of suspicion when new neurological deficits appear after central venous catheter manipulation, ensure meticulous preventive measures and close observation after removal, and rapidly initiate appropriate supportive treatment if venous air embolism is suspected.</p>