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Open access

Cronobacter brain abscess and refractory epilepsy in a newborn: role of epilepsy surgery. Illustrative case

Meredith Yang, John Tsiang, Melissa A. LoPresti, and Sandi Lam

BACKGROUND

Neonatal meningitis due to Cronobacter is associated with powdered infant formula. Prompt recognition of this rare but aggressive infection is critical.

OBSERVATIONS

The authors report a unique case of neonatal Cronobacter meningoencephalitis complicated by brain abscess and status epilepticus, requiring surgical intervention in a preterm 4-week-old male and related to contaminated powdered infant formula. They discuss the medical and surgical management in this patient, as well as the role of epilepsy surgery in acute drug-resistant epilepsy. This is paired with a literature review examining Cronobacter infections in infants to provide a summative review of the existing literature.

LESSONS

Cronobacter contamination in powdered infant formula and breast pumps is rare but can cause life-threatening infections. When evaluating patients with Cronobacter central nervous system infections, serial neuroimaging, infection control, and prompt surgical management are essential. Future studies are needed regarding the role of epilepsy surgery in the acute infectious period.

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

Vascular anomaly, lipoma, and polymicrogyria associated with schizencephaly: developmental and diagnostic insights. Illustrative case

Kevin K. Kumar, Angus Toland, Nancy Fischbein, Martha Morrell, Jeremy J. Heit, Donald E. Born, and Gary K. Steinberg

BACKGROUND

Schizencephaly is an uncommon central nervous system malformation. Intracranial lipomas are also rare, accounting for approximately 0.1% of brain “tumors.” They are believed to be derived from a persistent meninx primitiva, a neural crest–derived mesenchyme that develops into the dura and leptomeninges.

OBSERVATIONS

The authors present a case of heterotopic adipose tissue and a nonshunting arterial vascular malformation arising within a schizencephalic cleft in a 22-year-old male. Imaging showed right frontal gray matter abnormality and an associated suspected arteriovenous malformation with evidence of hemorrhage. Brain magnetic resonance imaging revealed right frontal polymicrogyria lining an open-lip schizencephaly, periventricular heterotopic gray matter, fat within the schizencephalic cleft, and gradient echo hypointensity concerning for prior hemorrhage. Histological assessment demonstrated mature adipose tissue with large-bore, thick-walled, irregular arteries. Mural calcifications and subendothelial cushions suggesting nonlaminar blood flow were observed. There were no arterialized veins or direct transitions from the arteries to veins. Hemosiderin deposition was scant, and hemorrhage was not present. The final diagnosis was consistent with ectopic mature adipose tissue and arteries with meningocerebral cicatrix.

LESSONS

This example of a complex maldevelopment of derivatives of the meninx primitiva in association with cortical maldevelopment highlights the unique challenges from both a radiological and histological perspective during diagnostic workup.

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

An epileptogenic intracranial cystic lesion lined with fallopian tube-type epithelium: illustrative case

Yuji Dekita, Yushin Takemoto, Kazutaka Ozono, Tadashi Hamasaki, Rin Yamada, Yoshiki Mikami, Jun-ichiro Kuroda, Nobuyuki Tsubota, and Akitake Mukasa

BACKGROUND

Intracranial cystic lesions are often a trigger for epileptic seizures. However, there has never been a report of a cystic lesion lined with fallopian tube-type epithelium.

OBSERVATIONS

A 48-year-old female presented with a cystic lesion in the right occipital lobe, which gradually grew over 8 years. Right occipital lobe epilepsy was diagnosed based on visual aura, convulsive seizures, and electroencephalogram findings and the cyst was surgically removed. Further examination revealed the cyst was lined with ciliated cells, which had morphological and immunohistochemical features similar to those of fallopian tube epithelium.

LESSONS

The characteristics of the cyst did not conform to any known types of benign cystic lesion. To the authors’ knowledge, no such cyst has been reported before. The authors discuss the origins and pathogenesis of this unfamiliar cystic lesion.

Open access

Magnetic resonance imaging–guided laser interstitial thermal therapy for complete corpus callosotomy: technique and 1-year outcomes. Patient series

Benjamin J. Best, Irene Kim, and Sean M. Lew

BACKGROUND

Magnetic resonance imaging (MRI)-guided stereotactic laser interstitial thermal therapy (LITT) is a minimally invasive technique that has been described for the treatment of certain forms of epilepsy through partial or complete callosotomy, with few cases describing single-stage complete LITT callosotomy. The authors aimed to demonstrate this technique’s feasibility and efficacy through description of the technique and 1-year outcomes in 3 cases of single-stage complete LITT callosotomy in patients with anatomically normal corpa callosa (CCs).

OBSERVATIONS

The patients were aged 14–27 years and experienced atonic seizures. Completeness of callosotomy was determined from MRI scans obtained >3 months after LITT procedures. The estimated ablations of the CC were 94%, 89%, and 100%, respectively. The second patient had a catheter breach the lateral ventricle, resulting in the lowest estimated percentage of ablation in this series (89%), with minimal atonic seizure reduction. The first patient had significant reduction in atonic seizure frequency, and the third patient had complete resolution of atonic seizures. None of the patients experienced any long-term complications. Intensive care length of stay was 1 night for each patient, and total length of stay was between 2 and 7 nights. Postoperative follow-up was between 14 and 18 months.

LESSONS

Complete laser callosotomy is achievable and is a safe alternative to microsurgical or endoscopic approaches.

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

Intact speech perception after resection of dominant hemisphere primary auditory cortex for the treatment of medically refractory epilepsy: illustrative case

Patrick W. Hullett, Nazineen Kandahari, Tina T. Shih, Jonathan K. Kleen, Robert C. Knowlton, Vikram R. Rao, and Edward F. Chang

BACKGROUND

In classic speech network models, the primary auditory cortex is the source of auditory input to Wernicke’s area in the posterior superior temporal gyrus (pSTG). Because resection of the primary auditory cortex in the dominant hemisphere removes inputs to the pSTG, there is a risk of speech impairment. However, recent research has shown the existence of other, nonprimary auditory cortex inputs to the pSTG, potentially reducing the risk of primary auditory cortex resection in the dominant hemisphere.

OBSERVATIONS

Here, the authors present a clinical case of a woman with severe medically refractory epilepsy with a lesional epileptic focus in the left (dominant) Heschl’s gyrus. Analysis of neural responses to speech stimuli was consistent with primary auditory cortex localization to Heschl’s gyrus. Although the primary auditory cortex was within the proposed resection margins, she underwent lesionectomy with total resection of Heschl’s gyrus. Postoperatively, she had no speech deficits and her seizures were fully controlled.

LESSONS

While resection of the dominant hemisphere Heschl’s gyrus/primary auditory cortex warrants caution, this case illustrates the ability to resect the primary auditory cortex without speech impairment and supports recent models of multiple parallel inputs to the pSTG.

Open access

Resection of an occipital lobe epileptogenic network resulting in improvement of a visual field deficit: illustrative case

Alper Dincer, John Herendeen, Joel Oster, and James Kryzanski

BACKGROUND

Drug-resistant epilepsy leads to significant morbidity and mortality. Epilepsy surgery for resection of seizure foci is underused, particularly when a seizure focus is located in eloquent cortex. Epileptogenic networks may lead to neurological deficits out of proportion to a causative lesion. Disruption of the network may lead not only to seizure freedom but also reversal of a neurological deficit.

OBSERVATIONS

A 32-year-old male with new-onset generalized tonic-clonic seizure was found to have an occipital lobe cavernous malformation. On visual field testing, he was found to have a right-sided hemianopsia. He did not tolerate antiepileptic drugs and had a significant decline in quality of life. Resection was planned using intraoperative electrocorticography to remove the cavernous malformation and disrupt the epileptogenic network. Immediate and delayed postoperative visual field testing demonstrated improvement of the visual field deficit, with near resolution of the deficit 6 weeks postoperatively.

LESSONS

Epilepsy networks in eloquent cortex may cause deficits that improve after the causative lesion is resected and the network disrupted, a concept that is underreported in the literature. A subset of patients with frequent epileptiform activity and preoperative deficits may experience postoperative neurological improvement along with relief of seizures.

Free access

Localizing seizure onset zones in surgical epilepsy with neurostimulation deep learning

Graham W. Johnson, Leon Y. Cai, Derek J. Doss, Jasmine W. Jiang, Aarushi S. Negi, Saramati Narasimhan, Danika L. Paulo, Hernán F. J. González, Shawniqua Williams Roberson, Sarah K. Bick, Catie E. Chang, Victoria L. Morgan, Mark T. Wallace, and Dario J. Englot

OBJECTIVE

In drug-resistant temporal lobe epilepsy, automated tools for seizure onset zone (SOZ) localization that use brief interictal recordings could supplement presurgical evaluations and improve care. Thus, the authors sought to localize SOZs by training a multichannel convolutional neural network on stereoelectroencephalography (SEEG) cortico-cortical evoked potentials.

METHODS

The authors performed single-pulse electrical stimulation in 10 drug-resistant temporal lobe epilepsy patients implanted with SEEG. Using 500,000 unique poststimulation SEEG epochs, the authors trained a multichannel 1-dimensional convolutional neural network to determine whether an SOZ had been stimulated.

RESULTS

SOZs were classified with mean sensitivity of 78.1% and specificity of 74.6% according to leave-one-patient-out testing. To achieve maximum accuracy, the model required a 0- to 350-msec poststimulation time period. Post hoc analysis revealed that the model accurately classified unilateral versus bilateral mesial temporal lobe seizure onset, as well as neocortical SOZs.

CONCLUSIONS

This was the first demonstration, to the authors’ knowledge, that a deep learning framework can be used to accurately classify SOZs with single-pulse electrical stimulation–evoked responses. These findings suggest that accurate classification of SOZs relies on a complex temporal evolution of evoked responses within 350 msec of stimulation. Validation in a larger data set could provide a practical clinical tool for the presurgical evaluation of drug-resistant epilepsy.