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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

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

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.

Open access

Removal of malformation in cerebral proliferative angiopathy: illustrative case

Gwang Yoon Choi, Hyuk Jai Choi, Jin Pyeong Jeon, Jin Seo Yang, Suk-Hyung Kang, and Yong-Jun Cho

BACKGROUND

Cerebral proliferative angiopathy (CPA) is a rare vascular disorder distinct from arteriovenous malformation. Because of the disorder’s rarity, there is still a controversy on the most promising treatment method for CPA. However, several meta-analysis articles suggest indirect vascularization such as encephalo-duro-arterio-synangiosis as an effective way of treating symptoms that are medically uncontrolled.

OBSERVATIONS

The authors describe a case of an 11-year-old boy with this disease, who had epilepsy that was intractable despite conservative management. The patient recovered from his symptoms after the vascular malformation was surgically removed. This is the first reported case of surgical removal in CPA.

LESSONS

Although further investigation on the best treatment for CPA is needed, the authors believe surgical intervention may also be an effective treatment modality when a patient presents with persisting symptoms.

Open access

Epileptogenic zone localization using intraoperative gamma oscillation regularity analysis in epilepsy surgery for cavernomas: patient series

Yosuke Sato, Yoshihito Tsuji, Yuta Kawauchi, Kazuki Iizuka, Yusuke Kobayashi, Ryo Irie, Tatsuya Sugiyama, and Tohru Mizutani

BACKGROUND

In epilepsy surgery for cavernoma with intractable focal epilepsy, removal of the cavernoma with its surrounding hemosiderin deposition and other extended epileptogenic zone has been shown to improve postsurgical seizures. However, there has been no significant association between such an epileptogenic zone and intraoperative electrocorticography (ECoG) findings. The authors recently demonstrated that high regular gamma oscillation (30–70 Hz) regularity (GOR) significantly correlates with epileptogenicity.

OBSERVATIONS

The authors evaluated the utility of intraoperative GOR analysis in epilepsy surgery for cavernomas. The authors also analyzed intraoperative ECoG data from 6 patients with cavernomas. The GOR was calculated using a sample entropy algorithm. In 4 patients, the GOR was significantly high in the area with the pathological hemosiderin deposition. In 2 patients with temporal cavernoma, the GOR was significantly high in both the hippocampus and the area with the pathological hemosiderin deposition. ECoG showed no obvious epileptic waveforms in 3 patients, whereas extensive spikes were observed in 3 patients. All patients underwent cavernoma removal plus resection of the area with significantly high GOR. The 2 patients with temporal cavernomas underwent additional hippocampal transection. All patients were seizure free after surgery.

LESSONS

The high GOR may be a novel intraoperative marker of the epileptogenic zone in epilepsy surgery for cavernomas.