Acta Neurochirurgica https://doi.org/10.1007/s00701-018-3606-9 CASE REPORT - FUNCTIONAL NEUROSURGERY - EPILEPSY Diffuse vasospasm after transcortical temporal lobectomy for intractable epilepsy James Charles Dickerson 1 1 1 & Joaquin Andres Hidalgo & Zachary Stidham Smalley & James Mason Shiflett 1 Received: 15 February 2018 / Accepted: 25 June 2018 # Springer-Verlag GmbH Austria, part of Springer Nature 2018 Abstract Cerebral delayed ischemia due to arterial vasospasm is a rare complication following epilepsy surgery. Here we report the third known case and first of diffuse vasospasm. A 48-year-old woman underwent a transcortical anterior left temporal lobectomy. Eleven days later, she had new-onset expressive aphasia with narrowing of the anterior, middle, and posterior cerebral arteries, and increased velocities via transcranial Doppler. She was treated with fluids, nimodipine, and permissive hypertension. At 6 months, her speech was near baseline. Cerebral vasospasm may represent a rare cause of morbidity after anterior temporal lobectomy; a literature review on the subject is presented. Keywords Epilepsy . Neurosurgery . Epilepsy surgery . Vasospasm . Diffuse . Triple H . Amygdalohippocampectomy . Temporal lobe resection . Transsylvian . Transcortical Abbreviations SAH Subarachnoid hemorrhage MRI Magnetic resonance imaging PET Positron emission tomography NSICU Neuroscience intensive care unit CT Computed tomography POD Post-operative day MCA Middle cerebral artery CTA Computed tomography angiography ICA Internal carotid artery This work has not been previously presented at any conference or published elsewhere. There was no clinical trial registration number for this work. This article is part of the Topical Collection on Functional Neurosurgery– Epilepsy * James Charles Dickerson jcdickerson@umc.edu ACA IV CSF Anterior cerebral artery Intravenous Cerebrospinal fluid Introduction Cerebral artery vasospasm is a known complication following subarachnoid hemorrhage (SAH), but has also been reported following a variety of neurosurgical procedures, including surgical resection for intractable epilepsy [1, 9, 10, 15, 19]. Presently, there are only two reported cases of clinically apparent vasospasm following epilepsy surgery, and both were limited to the ipsilateral vasculature [3, 12]. Here we report what we believe to be the first case of symptomatic diffuse vasospasm following an anterior temporal lobectomy. We discuss the clinical course, management, and relevant literature. Joaquin Andres Hidalgo jhidalgo@umc.edu Zachary Stidham Smalley zsmalley@umc.edu James Mason Shiflett jshiflett@umc.edu 1 Department of Neurosurgery, University of Mississippi Medical Center, 2500 North State Street, Jackson, MS 39216, USA Case presentation A 48-year-old right-handed African-American female suffering from complex partial seizures with secondary generalization was referred to our institution for cortical mapping and staged resection of epileptogenic foci. At the time of admission, she was experiencing manual and oromandibular automatisms with Acta Neurochir speech arrest approximately twice a week refractory to treatment with divalproex, lacosamide, and topiramate. Pre-operative Wada testing demonstrated language representation in the left hemisphere, and magnetic resonance imaging (MRI) without the administration of intravenous contrast showed left hippocampal sclerosis. Positron emission tomography (PET) scan did not reveal any abnormal hypermetabolic foci. Neuropsychological testing demonstrated no receptive or expressive language difficulties. She underwent a craniotomy for subdural electrode placement on hospital day 1, and video electrocorticography delineated an irritative zone in the lateral and proximal aspects of the left posterior temporal electrode corresponding to the left mesial temporal lobe. On hospital day 7, she was taken to the operating room for electrode removal and left temporal lobectomy using an anterior transcortical approach. Resection included the left temporal lobe, hippocampus, amygdala, and uncus; final pathology was consistent with type 1b cortical dysplasia. There were no intraoperative complications, and she was transferred to the neuroscience intensive care unit (NSICU) for 24-h post-operative observation. A routine post-operative MRI revealed bilateral temporal-occipital junction restricted diffusion, as well as right occipital horn and fourth ventricle interventricular hemorrhage (Fig. 1a, b). On post-operative day (POD) 1 following the lobectomy, the patient developed a fever and leukocytosis. Blood and urine cultures were drawn and then empiric broad spectrum antibiotics started; lower extremity ultrasonography was negative for thrombosis. On POD 4, a lumbar puncture was performed: the gram stain was negative but the white blood cell count was 12,792 with 90% neutrophils; the glucose, 12; and protein, 390. There were 46,727 red blood cells in the third tube. After 6 days, none of the cultures demonstrated growth, her fever and leukocytosis resolved, and the antibiotics were discontinued. Fig. 1 a Post-resection axial diffusion weighted MRI sequence showing parieto-occipital infarct. b Axial susceptibility weighted MRI sequence revealing right parieto-occipital cortical contusion and hemorrhage tract from deep subdural grid placement with subsequent interventricular hemorrhage in left occipital horn While the patient was intermittently lethargic after surgery, her mental status continued to improve following resolution of the fever and leukocytosis. On POD 11, she was noted to have new-onset expressive aphasia. She was transferred back to the NSICU and underwent an MRI, which revealed restricted diffusion in the left middle cerebral artery (MCA) distribution concerning for infarct. CT angiography (CTA) obtained the following day demonstrated severe diffuse narrowing of the distal internal carotid arteries (ICA), proximal anterior cerebral arteries (ACA), MCAs, and the entire posterior circulation (Fig. 2a). Transcranial Doppler showed peak flow velocities of 150 cm/s in the left ACA and 331 cm/s in the left MCA. On the right, velocities were 273 cm/s in ACA and 187 cm/s in the MCA. Table 1 shows all recorded peak velocities. Overall, vasospasm was most severe in the left MCA and right ACA territories based on transcranial Doppler (TCD). Treatment for vasospasm was initiated with fluids, nimodipine, and permissive hypertension. Eight days after the initial CTA, on POD 20, a follow-up CTA was obtained and demonstrated improvement in vasospasm (Fig. 2b). The patient was discharged to a swing bed and completed a 21-day course of nimodipine. At discharge, her aphasia was improving but was still present. At the 6-month follow-up visit, her speech was significantly improved, and she was near her pre-operative baseline with only mild dysnomia remaining. In addition, she remained seizure free on lacosamide and topiramate. Discussion Symptomatic vasospasm after epilepsy surgery is not an expected complication. This case represents only the third reported (Table 2), and as such our institution has no formalized protocol to screen patients post-operatively. Given the rarity of Acta Neurochir Fig. 2 a Axial CTA image showing diffuse narrowing of the cerebral circulation, notably in proximal left MCA territory. b Follow-up CTA 8 days later demonstrating increased diameter of the anterior and middle circulation compared to initial CTA in a the event, a low index of suspicion likely led to a delay in diagnosis. This may also have occurred in the case presented by Mandonett et al. (2009), where the patient’s aphasic symptoms worsened on POD 2, but the diagnosis did not occur until POD 5. This group also employed intravenous (IV) nimodipine and hydration and reported marked clinical improvement within 24 h, and symptom resolution within a few months [12]. In Chakravarty et al. (2015), the onset of symptoms occurred 38 h after the surgery, and the diagnosis appears to have been prompt. This patient received intraarterial nimodipine, as well as blood pressure augmentation, yielding symptom resolution within hours [3]. In the present case, it is possible that the delay in treatment contributed to the prolonged aphasia. While there was discussion of obtaining digital subtraction angiography after the onset of the aphasia, it was forgone as it was unlikely to change management. Another plausible explanation for the vasospasm in our patient is meningitis [2]. While no organism was isolated, and post-operative cerebrospinal fluid (CSF) studies must be interpreted with caution, the patients were suggestive of bacterial infection [16, 20]. Bacterial meningitis has been shown to lead to vasospasm, postulated to occur through cytokines such as IL-1β and IL-6 [5]. However, these cytokine levels are also Table 1 elevated in the CSF of rodents after trauma; simply undergoing an operation may result in an increase in IL-1β and IL-6 [22]. While these three cases of clinically apparent vasospasm following epilepsy surgery are the only examples known by the authors, previous works estimate that the incidence of radiographic vasospasm may be considerably higher. Schaller et al. (1998) defined vasospasm as flow velocity increases of > 50% in comparison to pre-operative TCDs. They found vasospasm in one or both of the MCAs in 14 of 20 patients (70%) undergoing amygdalohippocampectomy by the transsylvian approach, but notably all were asymptomatic [17]. In 2004, this same group demonstrated statistically significant increased flow velocities in the ipsilateral and contralateral MCAs relative to pre-operative baseline in the transsylvian approach (n = 40), and in the ipsilateral MCA alone in the transcortical approach (n = 40). For the ipsilateral MCA, the transsylvian group’s pooled peak velocity represented a 79% increase over pre-operative TCDs, while the transcortical group experienced a 49% increase. Therefore, it is likely that a significant portion of these patients met their previously used criteria for vasospasm. However, these increased velocities were not associated with neurologic deterioration [19]. Peak velocities by territory and laterality from all recorded transcranial Doppler examinations Post-operative day Left ACA (cm/s) Left MCA (cm/s) Lindegaard ratio (left) Right ACA (cm/s) Right MCA (cm/s) Lindegaard ratio (right) 13 14 17 18 20 21 150 120 176 134 148 189 331 265 293 326 326 296 9.66 6.19 8.36 11.1 8.57 8.38 273 248 169 287 287 271 187 225 234 194 173 167 3.25 4.41 3.92 4.24 4.10 4.00 Acta Neurochir Table 2 Comparison of reported cases of symptomatic vasospasm to date Publication Surgery Presenting symptoms Time from surgery to symptoms Treatment Outcome Mandonett et al. (2009) Antero-mesial temporal lobectomy Global aphasia 48 h IV nimodipine and hyperhydration Improvement within 24 h and complete resolution by 2 months Chakravarty et al. Selective (2015) amygdalohippocampectomy Contralateral weakness and global aphasia 38 h Intra-arterial nimodipine and noradrenaline infusion Present Case Expressive aphasia 12 days Permissive hypertension, IV fluids, and oral nimodipine Near-total return of power, speech, and recognition after 4 h Improvement within 10 days and near resolution by 6 months Anterior temporal lobectomy Lackner et al. (2012) defined vasospasm as velocities > 120 cm/s and found that 35 of 107 patients (33%) undergoing either selective amygdalohippocampectomy or temporal lobe resection had increased velocities. Eighteen had ipsilateral MCA vasospasm alone, and 17 were found to have bilateral MCA vasospasm [10]. The 35 patients with vasospasm had a significantly higher incidence of unexpected post-operative neurologic signs and symptoms, with the most common being headache, aphasia, and cranial nerve and/or extremity paresis. However, the study did not identify a timeline for these complications. It is unclear if they were the direct result of the increased velocities or simply unexpected post-operative complications that correlated with vasospasm found at a later date. Because of this, the clinical significance of the work remains unclear [7, 14]. This paper did identify female gender and a higher volume of post-operative blood as risk factors for vasospasm. The time from surgery to symptom onset in the three reported cases was remarkably different—38 h, 48 h, and 11 days. In SAH secondary to aneurysm rupture, vasospasm has been shown to occur over a wide range of 3–15 days, with a peak incidence around days 6–8 [4, 21]. In studies of post-traumatic vasospasm, there is also a large range (2–12 days), but with a peak incidence 2–4 days after the insult [15]. Given the different peak incidences, it is unclear if these represent distinct pathways, or convergent ones. In both aneurysmal and traumatic SAH, the volume of blood is predictive of vasospasm risk [4, 6, 13]. It is possible that the “micro-trauma” of surgery led to these cases of post-operative vasospasm, or that they occurred via the mechanism seen in aneurysmal SAH [18]. The pathophysiology of vasospasm after SAH is not well understood, and at present it is thought to be a product of the release of spasmogenic substances during blood clot lysis [8, 9]. Studies that have investigated vasospasm following surgery have speculated that it may be secondary to mechanical manipulation of the vasculature or due to the spillage of blood into the cisterns [1, 12, 18, 19]. In our transcortical approach, there was no direct manipulation of the ipsilateral MCA, nor would this alone account for the diffuse spasm. Given the lumbar puncture demonstrating blood in the subarachnoid space, we consider the degradation of blood products the more plausible mechanism. In Mandonett et al. (2009), subarachnoid blood was visualized on the initial post-operative scan. In Chakravarty et al. (2015), the post-operative scan was considered “unremarkable” and an image was not provided in the manuscript. It is possible that the volume of subarachnoid blood was larger in this case than in the other two, and this may have led to diffuse rather than local vasospasm. There was also intraventricular hemorrhage visualized on the postresection MRI, and vasospasm in association with intraventricular hemorrhage has been described [11]. It is possible that the burden of subarachnoid blood and intraventricular blood, and a possible meningitis, all contributed to an inflammatory state leading to diffuse vasospasm. Conclusion Here we report the third known case of symptomatic vasospasm after surgery for epilepsy, and the first known instance of diffuse vasospasm. Although rare, it is a documented and treatable cause of post-operative morbidity. Therefore, in the event of unexpected neurologic deficit post-resection, it is reasonable to consider vasospasm, especially in the setting of infection or the presence of subarachnoid or intraventricular blood. Compliance with ethical standards Conflict of interest The authors declare that they have no conflict of interest. Ethical approval This article does not contain any studies with human or animal participants performed by any of the authors. Patient consent The patient consented for the use of their case in scholarly activities, such as education and research. 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