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Douglas R. Taylor, Scott D. Wait, James W. Wheless and Frederick A. Boop

Neurocutaneous melanosis (NCM) is a rare, congenital neuroectodermal dysplasia generally resulting in early death from neurological decline due to leptomeningeal involvement. Nonmeningeal CNS epileptogenic lesions presenting in later childhood in the absence of leptomeningeal disease are rare. This report summarizes a rare presentation and curative epilepsy surgery.

The authors discuss the case of a 14-year-old girl with NCM who originally presented with intractable partial-onset seizures. The MRI, PET, and SPECT studies subsequently revealed a focal epileptogenic source in the right temporal lobe. Results of video-electroencephalography monitoring concurred with the imaging findings, and a right temporal lobectomy was performed including the medial structures. Following surgery, histopathological features of the lesion included multiple scattered mononuclear cells with brown pigmentation in the amygdala specimen. The patient remains seizure free 2 years postresection, and no longer needs medication for seizure management.

This patient presented with an atypical CNS manifestation of NCM that is curable by epilepsy surgery. Her intractable epilepsy developed secondary to amygdalar neuromelanosis, which had no associated leptomeningeal melanosis, an uncommon occurrence. As evidenced by the lack of seizure activity following resection, the patient's quality of life greatly improved after neurological surgery.

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Kristian Aquilina, Dave F. Clarke, James W. Wheless and Frederick A. Boop

Temporal lobe encephaloceles can be associated with temporal lobe epilepsy. The authors report on the case of an adolescent with multiple microencephaloceles, in the anterolateral middle fossa floor, identified at surgery (temporal lobectomy) for intractable partial-onset seizures of temporal origin. Magnetic resonance imaging revealed only hippocampal atrophy. Subdural electrodes demonstrated ictal activity arising primarily from the anterior and lateral temporal lobe, close to the microencephaloceles, spreading to the anterior and posterior mesial structures. Pathological examination revealed diffuse temporal gliosis involving the hippocampus, together with microdysgenesis of the amygdala. The literature on epilepsy secondary to encephaloceles is reviewed and the contribution of the microencephaloceles to the seizure disorder in this patient is discussed.

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Andrew C. Papanicolaou, Panagiotis G. Simos, Joshua I. Breier, George Zouridakis, L. James Willmore, James W. Wheless, Jules E. C. Constantinou, William W. Maggio and William B. Gormley

Object. In this paper the authors introduce a novel use of magnetoencephalography (MEG) for noninvasive mapping of language-specific cortex in individual patients and in healthy volunteers.

Methods. The authors describe a series of six experiments in which normative MEG data were collected and the reliability, validity, and topographical accuracy of the data were assessed in patients who had also undergone the Wada procedure or language mapping through intraoperative cortical stimulation.

Conclusions. Findings include: 1) receptive language—specific areas can be reliably activated by simple language tasks and this activation can be readily recorded in short MEG sessions; 2) MEG-derived maps of each individual are reliable because they remain stable over time and are independent of whether auditory or visual stimuli are used to activate the brain; and 3) these maps are also valid because they concur with results of the Wada procedure in assessing hemispheric dominance for language and with the results of cortical stimulation in identifying the precise topography of receptive language regions within the dominant hemisphere.

Although the MEG mapping technique should be further refined, it has been shown to be efficacious by correctly identifying the language-dominant hemisphere and specific language-related regions within this hemisphere. Further development of the technique may render it a valuable adjunct for routine presurgical planning in many patients who harbor tumors or have epilepsy.

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Andrew C. Papanicolaou, Panagiotis G. Simos, Joshua I. Breier, George Zouridakis, L. James Willmore, James W. Wheless, Jules E. C. Constantinou, William W. Maggio and William B. Gormley

Object

In this paper the authors introduce a novel use of magnetoencephalography (MEG) for noninvasive mapping of language-specific cortex in individual patients and in healthy volunteers.

Methods

The authors describe a series of six experiments in which normative MEG data were collected and the reliability, validity, and topographical accuracy of the data were assessed in patients who had also undergone the Wada procedure or language mapping through intraoperative cortical stimulation.

Conclusions

Findings include: 1) receptive language-specific areas can be reliably activated by simple language tasks and this activation can be readily recorded in short MEG sessions; 2) MEG-derived maps of each individual are reliable because they remain stable over time and are independent of whether auditory or visual stimuli are used to activate the brain; and 3) these maps are also valid because they concur with results of the Wada procedure in assessing hemispheric dominance for language and with the results of cortical stimulation in identifying the precise topography of receptive language regions within the dominant hemisphere.

Although the MEG mapping technique should be further refined, it has been shown to be efficacious by correctly identifying the language-dominant hemisphere and specific language-related regions within this hemisphere. Further development of the technique may render it a valuable adjunct for routine presurgical planning in many patients who harbor tumors or have epilepsy.

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Panagiotis G. Simos, Andrew C. Papanicolaou, Joshua I. Breier, James W. Wheless, Jules E. C. Constantinou, William B. Gormley and William W. Maggio

Object. In this paper the authors demonstrate the concordance between magnetic source (MS) imaging and direct cortical stimulation for mapping receptive language cortex.

Methods. In 13 consecutive surgical patients, cortex specialized for receptive language functions was identified noninvasively by obtaining activation maps aided by MS imaging in the context of visual and auditory word-recognition tasks. Surgery was then performed for treatment of medically intractable seizure disorder (eight patients), and for resection of tumor (four), or angioma (one). Mapping of language areas with cortical stimulation was performed intraoperatively in 10 patients and extraoperatively in three. Cortical stimulation mapping verified the accuracy of the MS imaging—based localization in all cases.

Conclusions. Information provided by MS imaging can be especially helpful in cases of atypical language representation, including bihemispheric representation, and location of language in areas other than those expected within the dominant hemisphere, such as the anterior portion of the superior temporal gyrus, the posteroinferior portion of the middle temporal gyrus, the basal temporal cortex, and the lateral temporooccipital cortex.

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Mark Van Poppel, James W. Wheless, Dave F. Clarke, Amy McGregor, Mark H. McManis, Freedom F. Perkins Jr., Katherine Van Poppel, Stephen Fulton and Frederick A. Boop

Object

Functional mapping is important for determining surgical candidacy and also in epilepsy surgery planning. However, in young children and uncooperative patients, language mapping has been particularly challenging despite the advances in performing noninvasive functional studies. In this study the authors review a series of children with epilepsy who underwent language mapping with magnetoencephalography (MEG) while sedated or sleeping, to determine receptive language localization for presurgical evaluation.

Methods

The authors undertook a retrospective review of patients who underwent MEG between December 2007 and July 2009, and identified 15 individuals who underwent passive language testing as part of their presurgical evaluation because they were unable to participate in traditional language testing, such as Wada or functional MRI. Factors necessitating passive language testing included age and neurocognitive development.

Results

Three of the 15 patients were deemed candidates for epilepsy surgery based on the results from standard preoperative testing, including video electroencephalography, MRI, and passive receptive language testing using MEG technology. The MEG studies were used successfully to localize language in all 3 patients, creating opportunities for seizure freedom through surgery that would not otherwise have been available. All 3 patients then underwent resective epilepsy surgery without experiencing postoperative language deficits.

Conclusions

This case series is the first to look at language mapping during sleep (passive language mapping) in which MEG was used and is the first to evaluate passive language testing in a patient population with intracranial pathological entities. This case series demonstrates that MEG can provide an alternative method for receptive language localization in patients with barriers to more traditional language testing, and in these 3 cases surgery was performed safely based on the results.

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Andrew C. Papanicolaou, Panagiotis G. Simos, Eduardo M. Castillo, Joshua I. Breier, Shirin Sarkari, Ekaterina Pataraia, Rebecca L. Billingsley, Scott Buchanan, James Wheless, Vijayalakshmi Maggio and William W. Maggio

Object. In this study the authors evaluated the sensitivity and selectivity of a noninvasive language mapping procedure based on magnetoencephalography (MEG), for determining hemispheric dominance for language functions.

Methods. Magnetic activation profiles of the brain were obtained from 100 surgical candidates (age range 8–56 years) with medically intractable seizure disorder by using a whole-head MEG system within the context of a word recognition task. The degree of language-specific activity was indexed according to the number of consecutive sources (modeled as single, moving current dipoles) in perisylvian brain areas. Only activity sources that were observed with a high degree of spatial and temporal overlap in two split-half data sets were used to compute the MEG laterality index. Independently, all patients underwent Wada testing for the determination of hemispheric dominance for language.

Independent clinical judgments based on MEG and Wada data showed a high degree of concordance (87%). Magnetoencephalography laterality judgments had an overall sensitivity of 98%, but a lower selectivity of 83%, which was due to the fact that MEG detected more activity in the nondominant hemisphere than was predicted based on the Wada test. A number of objective criteria were derived based on this large patient series to ensure data quality and bolster the clinical usefulness of MEG for language mapping.

Conclusions. Although the availability of MEG is still limited across epilepsy surgery centers, this study method may be substituted for the Wada procedure in assessing hemispheric dominance for language in select cases.

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Hideaki Ishibashi, Panagiotis G. Simos, Eduardo M. Castillo, William W. Maggio, James W. Wheless, Howard L. Kim, Vijay Venkataraman, Daniel K. Sanders, Joshua I. Breier, Wenbo Zhang, Robert N. Davis and Andrew C. Papanicolaou

Object. Magnetoencephalography (MEG) is a novel noninvasive diagnostic tool used to determine preoperatively the location of the epileptogenic zone in patients with epilepsy. The presence of focal slowing of activity recorded by electroencephalography (EEG) is an additional indicator of an underlying pathological condition in cases of intractable mesial temporal lobe epilepsy (MTLE). In the present study the authors examined the significance of focal, slow-wave and interictal spike activity detected using MEG in 29 patients who suffered from MTLE that was not associated with structural brain lesions.

Methods. All patients underwent resective surgery after MEG and EEG monitoring. Equivalent single-dipole modeling was applied to focal low-frequency magnetic activity (LFMA) and interictal paroxysmal activity. Lateralized LFMA was defined as trains of rhythmic activity over the temporal area, with frequencies lower than 7 Hz, which were easily distinguished from background activity.

Lateralized LFMA was found in 17 patients (58.6%); it always occurred on the side ipsilateral to the side of resection and displayed a maximum amplitude over the temporal area. Dipolar sources of magnetic flux computed during slow-wave trains were found in the majority of cases in the posterior superior temporal region and, occasionally, in mesial temporal structures that were subsequently resected. With respect to lateralization there was never disagreement between LFMA and MEG interictal spike sources. Thus, in patients with MTLE that is not associated with a mass lesion LFMA is topographically related to the epileptogenic area and, therefore, has value for reliable determination of the side and, possibly, the location of this area.

Conclusions. Although focal slowing of EEG background activity is generally considered to be a nonspecific sign of functional disturbance, interictal LFMA in patients with MTLE should be conceptualized as a distinct electrographic phenomenon that is directly related to the epileptogenic abnormality. Analyzing the interictal MEG distribution of LFMA and sharp activity improves the diagnostic utility of MEG in patients with suspected TLE who are undergoing surgical evaluation.