Epilepsy surgery in children with tuberous sclerosis complex: Presurgical evaluation and outcome

Susan Koh, Prasanna Jayakar, Catalina Dunoyer, Sharon E. Whiting, Trevor J. Resnick, Luis A. Alvarez, Glenn Morrison, John Ragheb, Antonio Prats, Patricia Dean, Samson B. Antel, Jamie Gilman, Michael S. Duchowny

Research output: Contribution to journalArticle

165 Citations (Scopus)

Abstract

Purpose: Children with tuberous sclerosis complex (TSC) benefit from excisional surgery if seizures can be localized to a single tuber. We evaluated the role of noninvasive studies to localize the epileptogenic tuber/region (ET/R) and the outcome of focal resection. Methods: We identified 21 children with TSC, ages 3 months to 15 years (mean 4.8 years). All had video-(electroencephalogram) EEG and magnetic resonance imaging (MRI) scans, and 18 also had ictal single photon emission-computed tomography (SPECT) studies. An ET/R was localized in 17 patients. Thirteen patients underwent resection guided by intraoperative electrocorticography (n = 7) or subdural monitoring (n = 6). Results: Interictal EEG revealed a principal spike focus (PSF) that corresponded to the ET/R in 14 children. In seven, PSFs occurred in rhythmic runs. PSFs were not observed remote from the ET/R. Focal polymorphic slowing and attenuation occurred in the region of the PSF in 11 patients. Sixteen patients demonstrated an ictal focus corresponding to the ET/R. Ictal SPECT revealed focal hyperperfusion correlating with the ET/R in 10 patients. Although the MRIs in all children revealed multiple tubers, the ET/R corresponded to a large discrete tuber in 8 patients and a calcified tuber in 13 patients. Patchy calcified tubers were also seen elsewhere in six patients. At a mean follow-up of 26 months, 9 of the 13 children who underwent surgery were seizure-free, one had greater than 75% reduction in seizures, two were unchanged, and one was lost to follow-up. New seizures developed in one child from a contralateral tuber. Conclusions: Surgical resection of an ET/R alleviates seizures in most children with TSC and intractable epilepsy. The scalp EEG and MRI help define the ET/R and improve case selection when ictal SPECT is nonlocalizing.

Original languageEnglish
Pages (from-to)1206-1213
Number of pages8
JournalEpilepsia
Volume41
Issue number9
StatePublished - Sep 25 2000

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Tuberous Sclerosis
Epilepsy
Seizures
Electroencephalography
Single-Photon Emission-Computed Tomography
Stroke
Magnetic Resonance Imaging
Lost to Follow-Up
Scalp

Keywords

  • Childhood epilepsy
  • Epilepsy surgery
  • Tuberous sclerosis

ASJC Scopus subject areas

  • Clinical Neurology
  • Neuroscience(all)

Cite this

Koh, S., Jayakar, P., Dunoyer, C., Whiting, S. E., Resnick, T. J., Alvarez, L. A., ... Duchowny, M. S. (2000). Epilepsy surgery in children with tuberous sclerosis complex: Presurgical evaluation and outcome. Epilepsia, 41(9), 1206-1213.

Epilepsy surgery in children with tuberous sclerosis complex : Presurgical evaluation and outcome. / Koh, Susan; Jayakar, Prasanna; Dunoyer, Catalina; Whiting, Sharon E.; Resnick, Trevor J.; Alvarez, Luis A.; Morrison, Glenn; Ragheb, John; Prats, Antonio; Dean, Patricia; Antel, Samson B.; Gilman, Jamie; Duchowny, Michael S.

In: Epilepsia, Vol. 41, No. 9, 25.09.2000, p. 1206-1213.

Research output: Contribution to journalArticle

Koh, S, Jayakar, P, Dunoyer, C, Whiting, SE, Resnick, TJ, Alvarez, LA, Morrison, G, Ragheb, J, Prats, A, Dean, P, Antel, SB, Gilman, J & Duchowny, MS 2000, 'Epilepsy surgery in children with tuberous sclerosis complex: Presurgical evaluation and outcome', Epilepsia, vol. 41, no. 9, pp. 1206-1213.
Koh S, Jayakar P, Dunoyer C, Whiting SE, Resnick TJ, Alvarez LA et al. Epilepsy surgery in children with tuberous sclerosis complex: Presurgical evaluation and outcome. Epilepsia. 2000 Sep 25;41(9):1206-1213.
Koh, Susan ; Jayakar, Prasanna ; Dunoyer, Catalina ; Whiting, Sharon E. ; Resnick, Trevor J. ; Alvarez, Luis A. ; Morrison, Glenn ; Ragheb, John ; Prats, Antonio ; Dean, Patricia ; Antel, Samson B. ; Gilman, Jamie ; Duchowny, Michael S. / Epilepsy surgery in children with tuberous sclerosis complex : Presurgical evaluation and outcome. In: Epilepsia. 2000 ; Vol. 41, No. 9. pp. 1206-1213.
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abstract = "Purpose: Children with tuberous sclerosis complex (TSC) benefit from excisional surgery if seizures can be localized to a single tuber. We evaluated the role of noninvasive studies to localize the epileptogenic tuber/region (ET/R) and the outcome of focal resection. Methods: We identified 21 children with TSC, ages 3 months to 15 years (mean 4.8 years). All had video-(electroencephalogram) EEG and magnetic resonance imaging (MRI) scans, and 18 also had ictal single photon emission-computed tomography (SPECT) studies. An ET/R was localized in 17 patients. Thirteen patients underwent resection guided by intraoperative electrocorticography (n = 7) or subdural monitoring (n = 6). Results: Interictal EEG revealed a principal spike focus (PSF) that corresponded to the ET/R in 14 children. In seven, PSFs occurred in rhythmic runs. PSFs were not observed remote from the ET/R. Focal polymorphic slowing and attenuation occurred in the region of the PSF in 11 patients. Sixteen patients demonstrated an ictal focus corresponding to the ET/R. Ictal SPECT revealed focal hyperperfusion correlating with the ET/R in 10 patients. Although the MRIs in all children revealed multiple tubers, the ET/R corresponded to a large discrete tuber in 8 patients and a calcified tuber in 13 patients. Patchy calcified tubers were also seen elsewhere in six patients. At a mean follow-up of 26 months, 9 of the 13 children who underwent surgery were seizure-free, one had greater than 75{\%} reduction in seizures, two were unchanged, and one was lost to follow-up. New seizures developed in one child from a contralateral tuber. Conclusions: Surgical resection of an ET/R alleviates seizures in most children with TSC and intractable epilepsy. The scalp EEG and MRI help define the ET/R and improve case selection when ictal SPECT is nonlocalizing.",
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T1 - Epilepsy surgery in children with tuberous sclerosis complex

T2 - Presurgical evaluation and outcome

AU - Koh, Susan

AU - Jayakar, Prasanna

AU - Dunoyer, Catalina

AU - Whiting, Sharon E.

AU - Resnick, Trevor J.

AU - Alvarez, Luis A.

AU - Morrison, Glenn

AU - Ragheb, John

AU - Prats, Antonio

AU - Dean, Patricia

AU - Antel, Samson B.

AU - Gilman, Jamie

AU - Duchowny, Michael S.

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N2 - Purpose: Children with tuberous sclerosis complex (TSC) benefit from excisional surgery if seizures can be localized to a single tuber. We evaluated the role of noninvasive studies to localize the epileptogenic tuber/region (ET/R) and the outcome of focal resection. Methods: We identified 21 children with TSC, ages 3 months to 15 years (mean 4.8 years). All had video-(electroencephalogram) EEG and magnetic resonance imaging (MRI) scans, and 18 also had ictal single photon emission-computed tomography (SPECT) studies. An ET/R was localized in 17 patients. Thirteen patients underwent resection guided by intraoperative electrocorticography (n = 7) or subdural monitoring (n = 6). Results: Interictal EEG revealed a principal spike focus (PSF) that corresponded to the ET/R in 14 children. In seven, PSFs occurred in rhythmic runs. PSFs were not observed remote from the ET/R. Focal polymorphic slowing and attenuation occurred in the region of the PSF in 11 patients. Sixteen patients demonstrated an ictal focus corresponding to the ET/R. Ictal SPECT revealed focal hyperperfusion correlating with the ET/R in 10 patients. Although the MRIs in all children revealed multiple tubers, the ET/R corresponded to a large discrete tuber in 8 patients and a calcified tuber in 13 patients. Patchy calcified tubers were also seen elsewhere in six patients. At a mean follow-up of 26 months, 9 of the 13 children who underwent surgery were seizure-free, one had greater than 75% reduction in seizures, two were unchanged, and one was lost to follow-up. New seizures developed in one child from a contralateral tuber. Conclusions: Surgical resection of an ET/R alleviates seizures in most children with TSC and intractable epilepsy. The scalp EEG and MRI help define the ET/R and improve case selection when ictal SPECT is nonlocalizing.

AB - Purpose: Children with tuberous sclerosis complex (TSC) benefit from excisional surgery if seizures can be localized to a single tuber. We evaluated the role of noninvasive studies to localize the epileptogenic tuber/region (ET/R) and the outcome of focal resection. Methods: We identified 21 children with TSC, ages 3 months to 15 years (mean 4.8 years). All had video-(electroencephalogram) EEG and magnetic resonance imaging (MRI) scans, and 18 also had ictal single photon emission-computed tomography (SPECT) studies. An ET/R was localized in 17 patients. Thirteen patients underwent resection guided by intraoperative electrocorticography (n = 7) or subdural monitoring (n = 6). Results: Interictal EEG revealed a principal spike focus (PSF) that corresponded to the ET/R in 14 children. In seven, PSFs occurred in rhythmic runs. PSFs were not observed remote from the ET/R. Focal polymorphic slowing and attenuation occurred in the region of the PSF in 11 patients. Sixteen patients demonstrated an ictal focus corresponding to the ET/R. Ictal SPECT revealed focal hyperperfusion correlating with the ET/R in 10 patients. Although the MRIs in all children revealed multiple tubers, the ET/R corresponded to a large discrete tuber in 8 patients and a calcified tuber in 13 patients. Patchy calcified tubers were also seen elsewhere in six patients. At a mean follow-up of 26 months, 9 of the 13 children who underwent surgery were seizure-free, one had greater than 75% reduction in seizures, two were unchanged, and one was lost to follow-up. New seizures developed in one child from a contralateral tuber. Conclusions: Surgical resection of an ET/R alleviates seizures in most children with TSC and intractable epilepsy. The scalp EEG and MRI help define the ET/R and improve case selection when ictal SPECT is nonlocalizing.

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KW - Epilepsy surgery

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