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Open access

Bilateral and asymmetrical localization of language function identified by the superselective infusion of propofol in an epilepsy patient with a mild malformation of cortical development: illustrative case

Mayuko Otomo, Shin-ichiro Osawa, Kyoko Suzuki, Kazuo Kakinuma, Kazushi Ukishiro, Hiroyoshi Suzuki, Kuniyasu Niizuma, Norio Narita, Nobukazu Nakasato, and Teiji Tominaga

BACKGROUND

Atypical localization of language function can result in unexpected postsurgical deficits after cortical resection, but it is difficult to predict the risk in the presurgical evaluation. The authors experienced a rare case of the bilateral and independent existence of different components of language function identified by segmented evaluation of anatomical anterior and posterior language areas using the superselective infusion of propofol.

OBSERVATIONS

A 32-year-old right-handed female presented with drug-resistant epilepsy. Comprehensive epilepsy evaluation suggested that the epileptic foci involved the whole left frontal lobe but provided less evidence of structural abnormality. To estimate the extent of functional deterioration likely to be caused by an extended left frontal lobectomy, the authors evaluated segmented cortical function in the ipsi- and contralateral hemispheres by the superselective infusion of propofol into the branches of the intracranial artery. The results revealed bilateral and asymmetrical localization of language function because the patient presented with different components of aphasia in each hemisphere. Based on the authors’ assessment of her functional tolerance, an extended left frontal lobectomy was performed and resulted in neurological deficits within the anticipated range.

LESSONS

An accurate understanding of the correlations between vascular and functional anatomy and the highly specific evaluation of language function provides more advanced presurgical assessment, allowing more tailored planning of cortical resection.

Open access

Stereoelectroencephalography in the setting of a previously implanted responsive neural stimulation device: illustrative case

Dorian M Kusyk, Nicholas Blaney, Timothy Quezada, and Alexander C Whiting

BACKGROUND

Responsive neural stimulation (RNS) is a relatively novel procedure for drug-resistant epilepsy, which involves implantation of a device into the skull and brain. As more devices are implanted, there may be an increasing need to perform intracranial electrocorticography in implant patients with persistent seizures. Given the device location, imaging difficulties with implanted devices, and other technical hurdles, stereoelectroencephalography (SEEG) could be especially challenging. The authors describe the first reported SEEG investigation in a patient with an RNS device, highlighting the technical challenges and clinical data ascertained.

OBSERVATIONS

A 41-year-old male with drug-resistant epilepsy presented several years after a local surgeon had placed an RNS device with two electrodes in the bilateral parieto-occipital lobes. Because of inadequate seizure control, the patient was offered a repeat SEEG investigation to characterize his epilepsy better. Although more technically challenging than a traditional SEEG implantation, the SEEG investigation was successfully performed, which led to a confirmation of bilateral hippocampal seizure onset. The patient underwent repositioning of his RNS leads with a significant decrease in his seizure frequency.

LESSONS

Concurrent implantation of SEEG electrodes in a functioning RNS device can be safely performed and can augment our understanding of a patient’s seizures.

Open access

A new strategy for treating drug-resistant focal aware seizures: thalamic specific nuclei deep brain stimulation. Illustrative case

Osvaldo Vilela-Filho, Hélio F. Silva-Filho, Lissa C. Goulart, Paulo C. Ragazzo, and Francisco M. Arruda

BACKGROUND

Focal aware seizures (FASs) are relatively common and frequently pharmaco-resistant. If the seizure onset zone (SOZ) is in eloquent cortical areas, making resective surgery risky and inadvisable, deep brain stimulation (DBS) of the anterior nucleus of the thalamus, which is efficacious in less than half of the cases, has been the main alternative. New targets should be searched to address this deficiency. The present study aims to determine if DBS of different thalamic specific nuclei can modulate the abnormal electrical activity of the SOZ located in their respective cortical projection areas. Herein, the authors present the first patient in an ongoing trial.

OBSERVATIONS

A 60-year-old female patient presented with 25-year history of pharmaco-resistant focal aware visual seizures frequently evolving to focal impaired awareness seizures. The SOZ was in the right occipital lobe (positron emission tomography-computed tomography/video electroencephalography). Magnetic resonance imaging was normal. She underwent ipsilateral lateral geniculate nucleus (LGN) DBS procedure. After a 24-month follow-up, seizure frequency decreased by 97%, improving quality of life and daily functioning without complications.

LESSONS

This is the first time the LGN has been targeted in humans. The results support the hypothesis that led to this study. This strategy represents a paradigm shift in the way of treating pharmaco-resistant FASs not amenable to resective surgery.

Open access

Radiofrequency ablation during stereoelectroencephalography: from diagnostic tool to therapeutic intervention. Illustrative case

Demitre Serletis, Juan Bulacio, Justin Bingaman, Elham Abushanab, Stephen P. Harasimchuk, Richard Rammo, Silvia Neme-Mercante, and William Bingaman

BACKGROUND

Radiofrequency thermocoagulation (RFTC) during intracranial stereoelectroencephalography (sEEG) was first described as a safe technique for creating lesions of epileptic foci in 2004. Since that time, the method has been applied as a diagnostic and/or palliative intervention. Although widely practiced in European epilepsy surgical programs, the technique has not been popularized in the United States given the lack of Food and Drug Administration (FDA)–approved technologies permitting safe usage of in situ sEEG electrodes for this purpose.

OBSERVATIONS

The authors present a case report of a young female patient with refractory left neocortical temporal lobe epilepsy undergoing sEEG electrode implantation, who underwent sEEG-guided RFTC via a stereotactic temperature-sensing pallidotomy probe. Although used as a diagnostic step in her workup, the patient has remained seizure-free for nearly 18 months.

LESSONS

The use of in situ sEEG electrodes for RFTC remains limited in the United States. In this context, this case highlights a safe alternative and temporizing approach to performing diagnostic sEEG-guided RFTC, using a temperature-sensing pallidotomy probe to create small, precise stereotactic lesions. The authors caution careful consideration of this technique as a temporary work-around solution while also highlighting the rising need for new FDA-approved technologies for safe RFTC through in situ temperature-sensing sEEG electrodes.

Open access

Focal drug-resistant temporal lobe epilepsy associated with an ipsilateral anterior choroidal artery aneurysm: illustrative case

H. Westley Phillips, Shivani D. Rangwala, Joanna Papadakis, David J. Segar, Melissa Tsuboyama, Anna L. R. Pinto, Joseph P. Harmon, Sulpicio G. Soriano, Carlos J. Munoz, Joseph R. Madsen, Alfred P. See, and Scellig S. Stone

BACKGROUND

The occurrence of both an intracranial aneurysm and epilepsy, especially drug-resistant epilepsy (DRE), is rare. Although the overall incidence of aneurysms associated with DRE is unclear, it is thought to be particularly infrequent in the pediatric population. Surgical ligation of the offending aneurysm has been reported in conjunction with resolving seizure activity, although few cases have cited a combined approach of aneurysm ligation and resection of an epileptogenic focus.

OBSERVATIONS

We present the case of a 14-year-old female patient with drug-resistant temporal lobe epilepsy and an ipsilateral supraclinoid internal carotid artery aneurysm. Seizure semiology, electroencephalography monitoring, and magnetic resonance imaging all indicated a left temporal epileptogenic focus, in addition to an incidental aneurysm. The authors recommended a combined surgery involving resection of the temporal lesion and surgical clip ligation of the aneurysm. Near-total resection and successful ligation were achieved, and the patient has remained seizure free since surgery at 1 year postoperatively.

LESSONS

In patients with focal DRE and an adjacent intracranial aneurysm, a combined surgical approach involving both resection and surgical ligation can be used. Several surgical timing and neuroanesthetic considerations should be made to ensure the overall safety and efficacy of this procedure.

Open access

Infraclavicular de novo placement of a responsive neurostimulator for a patient with eloquent glioma-associated epilepsy: illustrative case

Ahmad R. Masri, Bailey R. Yekzaman, Bradley J. Estes, Christopher S. Park, Patrick Landazuri, and Michael Kinsman

BACKGROUND

The authors present a 50-year-old female with high-grade glioma involving the motor cortex as the cause of her drug-resistant epilepsy (DRE). Responsive neurostimulation (RNS) was chosen for epilepsy treatment. Due to concerns regarding the generator impeding the regular imaging surveillance required for treatment and monitoring of her glioma, surgeons placed the internal pulse generator (IPG) within an infraclavicular chest pocket.

OBSERVATIONS

Implantation of the RNS device and IPG within the infraclavicular pocket was uneventful. However, both subdural and depth electrodes were used and connected to the IPG, and subdural electrodes are considerably shorter than depth electrodes (37 vs 44 cm). The shorter strip leads presumably generated significant tension, leading to fracture of the leads. Therefore, surgery was repeated using only depth electrodes for more length and less tension. The device has good-quality electrocorticography signals that continue to be used for device programming. The seizure burden was reduced, and quality of life improved for the patient.

LESSONS

The RNS system with infraclavicular IPG placement reduced the seizure burden and improved the quality of life of a patient with glioma-associated epilepsy. Surgeons may consider the infraclavicular location as an alternative site for implantation for RNS candidates who require recurrent intracranial magnetic resonance imaging.

Open access

Vasospasm secondary to responsive neurostimulator placement: a previously unreported complication. Illustrative case

Brandon Rogowski, Aaron Miller, Brian F. Saway, Jeffrey Wessell, Nathan C. Rowland, Jonathan Ross Lena, and William A. Vandergrift

BACKGROUND

The Responsive Neurostimulation (RNS) system is an implantable device for patients with drug-resistant epilepsy who are not candidates for resection of a seizure focus. As a relatively new therapeutic, the full spectrum of adverse effects has yet to be determined. A literature review revealed no previous reports of cerebral vasospasm following RNS implantation.

OBSERVATIONS

A 35-year-old man developed severe angiographic and clinical vasospasm following bilateral mesial temporal lobe RNS implantation. He initially presented with concerns for status epilepticus 8 days after implantation. On hospital day 3, a decline in his clinical examination prompted imaging studies that revealed a left middle cerebral artery (MCA) stroke with angiographic evidence of severe vasospasm of the left internal carotid artery (ICA), MCA, anterior cerebral artery (ACA), and right ICA and ACA. Despite improvements in angiographic vasospasm after appropriate treatment, a thrombus developed in the posterior M2 branch, requiring mechanical thrombectomy. Ultimately, the patient was stabilized and discharged to a rehabilitation facility with residual cognitive and motor deficits.

LESSONS

Cerebral vasospasm as a cause of ischemic stroke after uneventful RNS implantation is exceedingly rare, yet demands particular attention given the potential for severe consequences and the growing number of patients receiving RNS devices.

Open access

Dorsal medullary cavernous hemangioma presenting as obstinate hiccups and its surgical treatment: illustrative case

Sumirini Puppala, Abhijit Acharya, Atmaranjan Dash, and Surjyaprakash S. Choudhury

BACKGROUND

Hiccups are characterized by involuntary, intermittent, repetitive, myoclonic, and spasmodic contractions of the diaphragm. Hiccups are termed “intractable” when they last for over 1 month.

OBSERVATIONS

A rare case of intractable hiccups due to an uncommon location of cavernous hemangioma in the dorsal medulla is illustrated. With respect to the management, surgical excision was performed, and postsurgical complete recovery was witnessed, which has been reported only in six cases worldwide to date.

LESSONS

A mechanism of the hiccups reflex arc is discussed in detail with special reference to the need for equal emphasis on evaluating central nervous system causes and peripheral etiologies for pertinent hiccups.

Open access

Improved psychotic symptoms following resection of amygdalar low-grade glioma: illustrative case

John P. Andrews, Thomas A. Wozny, John K. Yue, and Doris D. Wang

BACKGROUND

Epilepsy-associated psychoses are poorly understood, and management is focused on treating epilepsy. Chronic, interictal psychosis that persists despite seizure control is typically treated with antipsychotics. Whether resection of a mesial temporal lobe lesion may improve interictal psychotic symptoms that persist despite seizure control remains unknown.

OBSERVATIONS

In a 52-year-old man with well-controlled epilepsy and persistent comorbid psychosis, brain magnetic resonance imaging (MRI) revealed an infiltrative, intraaxial, T2 fluid-attenuated inversion recovery intense mass of the left amygdala. The patient received an amygdalectomy for oncological diagnosis and surgical treatment of a presumed low-grade glioma. Pathology was ganglioglioma, World Health Organization grade I. Postoperatively, the patient reported immediate resolution of auditory hallucinations. Patient has remained seizure-free on 2 antiepileptic drugs and no antipsychotic pharmacotherapy and reported lasting improvement in his psychotic symptoms.

LESSONS

This report discusses improvement of psychosis symptoms after resection of an amygdalar glioma, independent of seizure outcome. This case supports a role of the amygdala in psychopathology and suggests that low-grade gliomas of the limbic system may represent, at minimum, partially reversible etiology of psychotic symptoms.

Open access

Laser ablative treatment of musicogenic epilepsy arising from dominant mesial temporal lobe: illustrative case

Christine Park, Saurabh R. Sinha, and Derek G. Southwell

BACKGROUND

Musicogenic epilepsy (ME) is a rare reflex epilepsy in which seizures are triggered by musical stimuli. Prior descriptions of ME have suggested localization to the nondominant temporal lobe, primarily in neocortex. Although resection has been described as a treatment for ME, other surgical modalities, such as laser ablation, may effectively disrupt seizure networks in ME while incurring comparatively lower risks of morbidity. The authors described the use of laser ablation to treat ME arising from the dominant mesial temporal structures.

OBSERVATIONS

A 37-year-old woman with a 15-year history of drug-resistant ME was referred for surgical evaluation. Her seizures were triggered by specific musical content and involved behavioral arrest, repetitive swallowing motions, and word incomprehension. Diagnostic studies, including magnetic resonance imaging, single-photon emission computed tomography, magnetoencephalography, Wada testing, and stereoelectroencephalography, indicated seizure onset in the left (dominant) mesial temporal lobe. Laser interstitial thermal therapy was used to ablate the left mesial seizure onset zone. The patient was discharged on postoperative day two. At 18-month follow-up, she was seizure-free with no posttreatment neurological deficits.

LESSONS

Laser ablation can be an effective treatment option for well-localized forms of ME, particularly when seizures originate from the dominant mesial temporal lobe.