BRAIN and NERVE－神経研究の進歩67巻4号

2015年04月発行

原著

胚芽異形成性上皮腫瘍に伴う難治性てんかん症例における発作起始域の臨床的・病理学的検討

著者： 村上信哉¹ 森岡隆人² 橋口公章¹ 鈴木諭³ 重藤寛史⁴ 酒田あゆみ⁵ 佐々木富男¹

所属機関： ¹九州大学大学院医学研究院脳神経外科 ²九州労災病院脳神経外科 ³九州大学大学院医学研究院神経病理学 ⁴九州大学大学院医学研究院神経内科 ⁵九州大学病院中央検査部脳波室

ページ範囲：P.525 - P.532

文献概要

胚芽異形成性上皮腫瘍（DNT）のてんかん原性はいまだに議論の多いところであるが，DNTのてんかん原性は病理学的に合併する皮質形成異常に存在するという考え方が主流である。九州大学脳神経外科で行ったてんかん原性域としてDNTがみられた難治性てんかんの手術3例のうち，2例は慢性硬膜下電極記録を経由して手術を行い，1例は術中皮質電位記録を用いて手術を行った。慢性硬膜下電極記録を用いた2例とも，DNTの周囲皮質に発作起始域を同定し，これらを含めて切除し，良好な発作転帰を得た。しかし，同部位の病理学的検索では，軽度のグリオーシスがみられるのみで，皮質形成異常は合併していなかった。術中皮質電位記録を用いた側頭葉外側部DNTの1例では，側頭葉内側部に脳波異常域を同定したが，腫瘍を含めた側頭葉外側部を切除し，良好な発作転帰は得られなかった。DNT症例のてんかん外科における摘出範囲は，腫瘍の摘出だけでよいという考え方と，周囲皮質を含めて追加切除したほうがよいという考え方があるが，本報告のようにDNTのてんかん原性は症例によりさまざまであると考えられるので，慢性硬膜下電極記録を中心とした詳細な術前検査により正確なてんかん原性域の同定が重要である。

参考文献

1) Daumas-Duport C, Scheithauer BW, Chodkiewicz JP, Laws ER Jr, Vedrenne C: Dysembryoplastic neuroepithelial tumor: a surgically curable tumor of young patients with intractable partial seizures: report of thirty-nine cases. Neurosurgery 23: 545-556, 1988

2) Luders HO, Engel J Jr, Munari C: General principles. Engel J Jr(ed): Surgical Treatment of the Epilepsies, 2nd ed. Raven Press, New York, 1998, pp137-153

3) Blumcke I, Thom M, Aronica E, Armstrong DD, Vinters HV, et al: The clinicopathologic spectrum of focal cortical dysplasias: a consensus classification proposed by an ad hoc Task Force of the ILAE Diagnostic Methods Commission. Epilepsia 52: 158-174, 2011

4) Daumas-Duport C, Varlet P, Bacha S, Beuvon F, Cervera-Pierot P, et al: Dysembryoplastic neuroepithelial tumors: nonspecific histological forms: a study of 40 cases. J Neurooncol 41: 267-280, 1999

5) Honavar M, Janota I, Polkey CE: Histological heterogeneity of dysembryoplastic neuroepithelial tumour: identification and differential diagnosis in a series of 74 cases. Histopathology 34: 342-356, 1999

6) Lee DY, Chung CK, Hwang YS, Choe G, Chi JG, et al: Dysembryoplastic neuroepithelial tumor: radiological findings (including PET, SPECT, and MRS) and surgical strategy. J Neurooncol 47: 167-174, 2000

7) Prayson RA, Estes ML, Morris HH: Coexistence of neoplasia and cortical dysplasia in patients presenting with seizures. Epilepsia 34: 609-615, 1993

8) Prayson RA, Fong J, Najm I: Coexistent pathology in chronic epilepsy patients with neoplasms. Mod Pathol 23: 1097-1103, 2010

9) Sakuta R, Otsubo H, Nolan MA, Weiss SK, Hawkins C, et al: Recurrent intractable seizures in children with cortical dysplasia adjacent to dysembryoplastic neuroepithelial tumor. J Child Neurol 20: 377-384, 2005

10) Sharma MC, Jain D, Gupta A, Sarkar C, Suri V, et al: Dysembryoplastic neuroepithelial tumor: a clinicopathological study of 32 cases. Neurosurg Rev 32: 161-169, 2009

11) Takahashi A, Hong SC, Seo DW, Hong SB, Lee M, et al: Frequent association of cortical dysplasia in dysembryoplastic neuroepithelial tumor treated by epilepsy surgery. Surg Neurol 64: 419-427, 2005

12) Aronica E, Leenstra S, van Veelen CW, van Rijen PC, Hulsebos TJ, et al: Glioneuronal tumors and medically intractable epilepsy: a clinical study with long-term follow-up of seizure outcome after surgery. Epilepsy Res 43: 179-191, 2001

13) Minkin K, Klein O, Mancini J, Lena G: Surgical strategies and seizure control in pediatric patients with dysembryoplastic neuroepithelial tumors: a single-institution experience. J Neurosurg Pediatr 1: 206-210, 2008

14) Kameyama S, Fukuda M, Tomikawa M, Morota N, Oishi M, et al: Surgical strategy and outcomes for epileptic patients with focal cortical dysplasia or dysembryoplastic neuroepithelial tumor. Epilepsia 42 (Suppl 6): 37-41, 2001

15) Lee J, Lee BL, Joo EY, Seo DW, Hong SB, et al: Dysembryoplastic neuroepithelial tumors in pediatric patients. Brain Dev 31: 671-681, 2009

16) Morioka T, Hashiguchi K, Nagata S, Miyagi Y, Yoshida F, et al: Additional hippocampectomy in the surgical management of intractable temporal lobe epilepsy associated with glioneuronal tumor. Neurol Res 29: 807-815, 2007

17) Hashiguchi K, Morioka T, Yoshida F, Miyagi Y, Nagata S, et al: Correlation between scalp-recorded electroencephalographic and electrocorticographic activities during ictal period. Seizure 16: 238-247, 2007

FDG-positron emission tomography in medial temporal lobe epilepsy. Neurol Res 29: 215-222, 2007

19) Hashiguchi K, Morioka T, Murakami N, Suzuki SO, Hiwatashi A, et al: Utility of 3-T FLAIR and 3D short tau inversion recovery MR imaging in the preoperative diagnosis of hippocampal sclerosis: direct comparison with 1.5-T FLAIR MR imaging. Epilepsia 51: 1820-1828, 2010

I-iomazenil SPECT: a comparison with IMP SPECT and FDG PET. Nucl Med Commun 27: 893-899, 2006

21) Raymond AA, Fish DR, Sisodiya SM, Alsanjari N, Stevens JM, et al: Abnormalities of gyration, heterotopias, tuberous sclerosis, focal cortical dysplasia, microdysgenesis, dysembryoplastic neuroepithelial tumour and dysgenesis of the archicortex in epilepsy: clinical, EEG and neuroimaging features in 100 adult patients. Brain 118 (Pt 3): 629-660, 1995

22) Mikuni N, Ikeda A, Takahashi JA, Nozaki K, Miyamoto S, et al: A step-by-step resection guided by electrocorticography for nonmalignant brain tumors associated with long-term intractable epilepsy. Epilepsy Behav 8: 560-564, 2006

23) Kanata A, Morioka T, Tsukamoto H, Katsuta T, Suzuki SO: A patient with a dysembryoplastic neuroepithelial tumor who underwent epilepsy surgery after initial seizure. Pediatr Neurosurg 47: 436-441, 2011

24) Avoli M, Louvel J, Pumain R, Kohling R: Cellular and molecular mechanisms of epilepsy in the human brain. Prog Neurobiol 77: 166-200, 2005

25) Alonso-Nanclares L, De Felipe J: Vesicular glutamate transporter 1 immunostaining in the normal and epileptic human cerebral cortex. Neuroscience 134: 59-68, 2005

26) Aronica E, Yankaya B, Jansen GH, Leenstra S, van Veelen CW, et al: Ionotropic and metabotropic glutamate receptor protein expression in glioneuronal tumours from patients with intractable epilepsy. Neuropathol Appl Neurobiol 27: 223-237, 2001

27) Wolf HK, Roos D, Blumcke I, Pietsch T, Wiestler OD: Perilesional neurochemical changes in focal epilepsies. Acta Neuropathol 91: 376-384, 1996

28) Aronica E, Redeker S, Boer K, Spliet WG, van Rijen PC, et al: Inhibitory networks in epilepsy-associated gangliogliomas and in the perilesional epileptic cortex. Epilepsy Res 74: 33-44, 2007

I］-iomazenil-SPECT in localising the epileptogenic cortex. J Neurol Neurosurg Psychiatry 63: 743-748, 1997

30) Aubert S, Wendling F, Regis J, McGonigal A, Figarella-Branger D, et al: Local and remote epileptogenicity in focal cortical dysplasias and neurodevelopmental tumours. Brain 132: 3072-3086, 2009

31) Lee MC, Kang JY, Seol MB, Kim HS, Woo JY, et al: Clinical features and epileptogenesis of dysembryoplastic neuroepithelial tumor. Childs Nerv Syst 22: 1611-1618, 2006

32) Morioka T, Nishio S, Ishibashi H, Muraishi M, Hisada K, et al: Intrinsic epileptogenicity of focal cortical dysplasia as revealed by magnetoencephalography and electrocorticography. Epilepsy Res 33: 177-187, 1999