Cerebral infarction following cyanoacrylate endoscopic therapy of duodenal varices in a patient with a patent foramen ovale: comment

Abstract

We read with great interest the article by Abbar et al. [1] published in the journal. We recently also managed a case of cerebral infarction following N-butyl-2-cyanoacrylate glue sclerotherapy of esophageal varices in a patent with a previously unknown foramen ovale. The simultaneous presence of these conditions is quite rare as to our knowledge only two cases [1, 2] have been reported in the literature so far. This clinical scenario involves the challenging management of multiple concomitant thrombotic and hemorrhagic risks factors. Here, we report the case of an 18-year-old man with no relevant past medical history admitted to the Emergency Unit of our Hospital because of an episode of massive hematemesis. He underwent an urgent upper-GI endoscopy that showed oesophageal varices F3 with no signs of active bleeding and an actively bleeding gastroesophageal varix (GOV) type I, which was successfully treated with 4 mL N-butyl-2-cyanoacrylate injection. A subsequent abdominal CT scan showed a portal cavernomatosis and spleno-renal shunt, with no radiological signs of cirrhosis. Less than 24 h after the endoscopic procedure, the patient suddenly developed global aphasia and right hemiparesis. Brain CT and MRI scans showed a hyperdense focus of the M2 segment (left middle cerebral artery) (Fig. 1) and a recent onset insular ischemic lesion, with no significant ischemic penumbra. Given the recent massive bleeding and the high risk of re-bleeding, thrombolysis was not performed, while mechanical thrombectomy was not executed due to the absence of significant ischaemic penumbra. Nevertheless, neurological symptoms regressed within the next 24 h and the patient presented no residual neurological signs. Continuous ECG monitoring during observation in ICU documented persistent normal sinus rhythm with no phase of atrial fibrillation. Transthoracic echocardiogram and transcranial Doppler bubble-test were performed, to clarify the cause of cerebral infarction: the first showed minimal bulging of the atrial septum without septal aneurysm and no other abnormalities, (in particular normal atrium volume, no valvular abnormalities and normal systolic function), while the latter demonstrated a mild right-to-left shunt after the Valsalva maneuver (five bubbles after Valsalva), indicating the presence of a patent foramen ovale (PFO). Transoesophageal echocardiogram could not be performed due to the recent variceal ligation. No active sources of emboli were identified on subsequent ultrasound examinations. Carotid arteries were normal, no signs of deep vein thrombosis were documented at upper and lower limbs. A venous and cardio-embolic origin of the event was excluded and a correlation between endoscopic procedure and cerebral ischemic event was supposed, with cyanoacrylate glue as the probable cause of paradoxical embolism in a patient with confirmed PFO. Cyanoacrylate cerebral embolization is a rare occurrence, but the neuroradiological presentation is not atypical [3] so the diagnosis remains one of exclusion. In addition, thrombophilia screening showed inherited Protein C deficiency (activity 27%). Given the varices-associated haemorrhagic risk, several endoscopic band ligation procedures were performed to obtain varices eradication. It was then possible to start anticoagulant therapy with vitamin K antagonists, in consideration of the inherited high thrombotic risk. No further thrombotic and bleeding episode occurred at 12-month follow-up. * Giuseppe Marrone [email protected]