Case report: Capgras syndrome: a clinical manifestation of watershed cerebral infarct complicating the use of extracorporeal membrane oxygenation

Critical Care August 2001 Vol 5 No 4 Dejode et al Case report Capgras syndrome: a clinical manifestation of watershed cerebral infarct complicating the use of extracorporeal membrane oxygenation Jean Marc Dejode, François Antonini, Pierre Lagier and Claude Martin Department of Anesthesia and Intensive Care, CHU Nord, Marseilles, France Correspondence: Jean-Marc Dejode, Received: 16 May 2000 Revisions requested: 20 November 2000 Revisions received: 23 May 2001 Critical Care 2001, 5:232–235 Accepted: 4 June 2001 Published: 13 July 2001 © 2001 Dejode et al, licensee BioMed Central Ltd (Print ISSN 1364-8535; Online ISSN 1466-609X) This article is online at http://ccforum.com/content/5/4/232 Abstract Ischaemic cerebral accidents are frequent following extracorporeal membrane oxygenation (ECMO), especially after fixing the reinjection cannula in the right primitive carotid artery, which leads to an interruption in downstream flow. We describe a rare and unusual symptom of cerebral ischaemic accident that is known as Capgras syndrome. This feature is interesting because it may be documented by computed tomography (CT) scan and particular electroencephalography signals. It appears that our observation represents the first documented case of Capgras syndrome complicating ECMO. This incident emphasizes the potential hazards associated with right common artery ligature for venoarterial extracorporeal membrane oxygenation (VAECMO). In addition, it shows that this psychiatric symptom (that has been interpreted psychodynamically for many years) can have an organic basis, which should be studied. Keywords Capgras syndrome, cerebrovascular lesion, extracorporeal membrane oxygenation (ECMO), illusion of doubles, periodic lateralized epileptiform discharge (PLED) Introduction Watershed cerebral infarcts are very rare ischaemic lesions [1]. In the present report, we describe the case of a 24-year-old woman who developed border zone infarction after right primitive carotid artery occlusion for VAECMO. Case report A 24-year-old-woman was admitted to the emergency service of our hospital with severe adult respiratory distress syndrome, complicating pneumococcal pneumonia. Body temperature was 41.7°C, and blood cultures and bronchial samples were positive for Streptococcus pneumoniae. The patient was intubated and ventilated because of serious hypoxaemia. Mechanical ventilation parameters needed to be modified regularly, and pressures were gradually increased in order to combat hypoxaemia. This resulted in an aggressive assisted ventilation: fractional inspired oxygen 1; positive end-expiratory pressure 20 cmH2O; mean airway pressure 40 cmH2O; and inspiratory : expiratory ratio 2 : 1. Despite this aggressive approach, oxygenation remained insufficient: arterial oxygen saturation 65%; arterial oxygen tension 44 mmHg; arterial carbon dioxide tension 56 mmHg; and alveolar–arterial oxygen difference 600 mmHg. Nitric oxide inhalation (20 parts/million), in combination with almitrine adminstration, resulted in no improvement. Major air leak syndrome occurred, with pneumomediastinum, pneumoperitoneum and subcutaneous emphysema. CT = computed tomography; ECMO = extracorporeal membrane oxygenation; ICA = internal carotid artery; PLED = periodic lateralized epileptiform discharge; VAECMO = venous arterial extracorporeal membrane oxygenation. Available online http://ccforum.com/content/5/4/232 Figure 1 commentary review Periodic lateralized epileptiform discharges (C4-O2). Electroencephalography showed periodic lateralized epileptiform discharges (PLEDs) in the posterior right hemisphere (channel C4-O2; Fig. 1). Angiography, performed after surgical repair (on day 36 after admission), revealed normal flow in the repaired carotid artery and in the circle of Willis. CT scan revealed a posterior water- meeting abstracts Confronted with this hypoxic multiple organ failure, we opted to initiate VAECMO treatment, which was started at 45 h after admission. Blood was collected from the right atrium by a drain CH24, and reinjected using a Harvey’s cannula no 22 in the right primitive carotid artery following ligature of the downstream segment. The membrane used was a Ultrox I (3.5 m2) (Avecoz Cardiovascular France, Voisins le Bretonneux, France), the rate of carbon dioxide removal was 160 ml/min, and arterial oxygen saturation was above 96%. Apneic ventilation was started with a respiratory frequency of 4 breaths/min and a mean airway pressure of 16 cmH2O. Rapid improvements in the air leak syndrome and in the multiple organ failure (heart, kidney, liver) were observed within a few hours after beginning ECMO. On day 29 after admission epileptic fits with myoclonic movements of the right lower limb occurred. No precipitating factors (hypotension, hypoxia, electrolyte disorders) could be identified to explain the seizures. On clinical examination left hemiparesis was found. The patient complained of optical illusions, with permanent distortion of objects, and she thought that some intensive care unit physicians had been replaced by a double (identical looking imposter). She stated that there were true and false physicians. She was quite conscious of the abnormality of these phenomena, and there were no psychiatric antecedents. Thus, a diagnosis of Capgras syndrome was made. research Renal failure occurred, with anuria and fluid overload: natremia 129 mmol/l; total protein 34 g/l; blood nitrogen 21 mmol/l; and creatininaemia 350 µmol/l. Hepatic failure was present, with bilirubin at 120 µmol/l and a prothrombin time of 30 s. On day 10 after admission, VAECMO was stopped because of disseminated intravascular coagulation. Standard mechanical ventilation was then reinitiated. Haematosis and vital functions were maintained. On day 12 following admission, reconstruction of the carotid artery was performed, with an internal saphenous vein graft. We began to reduce sedation on day 22 after admission, and the patient progressively regained normal consciousness. At that time neurological examination findings were normal. reports The patient was administered noradrenaline (norepinephrine; 0.4 µg/kg per min), dopamine (15 µg/kg per min) and dobutamine (10 µg/kg per min), and the following measures were obtained: mean arterial pressure 50 mmHg; cardiac index 2.26 l/min per m2; systemic vascular resistance 659 dyne×s/cm5 per m2; central venous pressure 19 mmHg; mean pulmonary artery pressure 32 mmHg; pulmonary capillary wedge pressure 23 mmHg; and pulmonary vascular resistance 191 dyne×s/cm5 per m2. Critical Care August 2001 Vol 5 No 4 Dejode et al Figure 2 CT scan: watershed cerebral infarct between the right middle and the right posterior cerebral arterial territories. shed cerebral infarct (between the right middle and the right posterior cerebral arterial territories; Fig. 2). Anticonvulsant therapy was initiated. The epileptic fits declined progressively, and then stopped. The signs o (...truncated)