Neuropediatrics 2018; 49(03): 173-179
DOI: 10.1055/s-0038-1645871
Original Article
Georg Thieme Verlag KG Stuttgart · New York

Surgical Treatment and Long-Term Outcome of Cerebral Cavernous Malformations-Related Epilepsy in Pediatric Patients

Qiao Lin
1   Department of Neurosurgery, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
,
Peng-Fan Yang
1   Department of Neurosurgery, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
,
Yan-Zeng Jia
2   Department of Epileptology, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
,
Jia-Sheng Pei
1   Department of Neurosurgery, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
,
Hui Xiao
3   Department of Medical Imaging, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
,
Ting-Ting Zhang
4   Department of Pathology, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
,
Zhong-Hui Zhong
2   Department of Epileptology, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
,
Shou-Sen Wang
1   Department of Neurosurgery, Fuzhou General Hospital, Fujian Medical University, Fuzhou, China
› Author Affiliations
Further Information

Publication History

10 August 2017

01 March 2018

Publication Date:
20 April 2018 (online)

Abstract

Cerebral cavernous malformations (CMs) are vascular malformations affecting any part of the central nervous system. Clinical data and surgical outcomes for 27 pediatric patients with CM-related epilepsy were retrospectively reviewed. The mean age of onset was 12.71 ± 4.09 years, and the mean duration of epilepsy was 2.34 ± 1.95 years. All 27 patients were treated with microsurgery for resection of the CMs, and the hemosiderin rim, and the secondary epileptogenic zone if necessary. The mean follow-up period was 6.34 ± 3.35 years, and the overall postoperative outcomes were positive. Note that 77.8% of patients were seizure-free postoperatively. The other patients with residual epilepsy received incomplete resection of the hemosiderin rim or the secondary epileptogenic zone due to retention of vital neurological functions. Surgical treatment for pediatric patients with symptomatic supratentorial CMs is safe and effective. Early intervention is recommended to resect CMs, the hemosiderin rim, and the epileptogenic cortex, even in cases of multiple CMs.

 
  • References

  • 1 Lim M, Haddix T, Harsh GR, Vogel H, Steinberg GK, Guccione S. Characterization of the integrin alpha v beta3 in arteriovenous malformations and cavernous malformations. Cerebrovasc Dis 2005; 20 (01) 23-27
  • 2 Seker A, Yildirim O, Kurtkaya O. , et al. Expression of integrins in cerebral arteriovenous and cavernous malformations. Neurosurgery 2006; 58 (01) 159-168 , discussion 159–168
  • 3 Del Curling Jr O, Kelly Jr DL, Elster AD, Craven TE. An analysis of the natural history of cavernous angiomas. J Neurosurg 1991; 75 (05) 702-708
  • 4 Robinson JR, Awad IA, Little JR. Natural history of the cavernous angioma. J Neurosurg 1991; 75 (05) 709-714
  • 5 Mottolese C, Hermier M, Stan H. , et al. Central nervous system cavernomas in the pediatric age group. Neurosurg Rev 2001; 24 (2-3): 55-71 , discussion 72–73
  • 6 Maraire JN, Awad IA. Intracranial cavernous malformations: lesion behavior and management strategies. Neurosurgery 1995; 37 (04) 591-605
  • 7 Scott RM, Barnes P, Kupsky W, Adelman LS. Cavernous angiomas of the central nervous system in children. J Neurosurg 1992; 76 (01) 38-46
  • 8 Lee JW, Kim DS, Shim KW. , et al. Management of intracranial cavernous malformation in pediatric patients. Childs Nerv Syst 2008; 24 (03) 321-327
  • 9 Acciarri N, Galassi E, Giulioni M. , et al. Cavernous malformations of the central nervous system in the pediatric age group. Pediatr Neurosurg 2009; 45 (02) 81-104
  • 10 Cossu M, Raneri F, Casaceli G, Gozzo F, Pelliccia V, Lo Russo G. Surgical treatment of cavernoma-related epilepsy. J Neurosurg Sci 2015; 59 (03) 237-253
  • 11 Josephson CB, Rosenow F, Al-Shahi Salman R. Intracranial vascular malformations and epilepsy. Semin Neurol 2015; 35 (03) 223-234
  • 12 Di Rocco C, Iannelli A, Tamburrini G. Cavernomas of the central nervous system in children. A report of 22 cases. Acta Neurochir (Wien) 1996; 138 (11) 1267-1274 , discussion 1273–1274
  • 13 von der Brelie C, Kuczaty S, von Lehe M. Surgical management and long-term outcome of pediatric patients with different subtypes of epilepsy associated with cerebral cavernous malformations. J Neurosurg Pediatr 2014; 13 (06) 699-705
  • 14 Hugelshofer M, Acciarri N, Sure U. , et al. Effective surgical treatment of cerebral cavernous malformations: a multicenter study of 79 pediatric patients. J Neurosurg Pediatr 2011; 8 (05) 522-525
  • 15 Engel Jr J. Classification of epileptic disorders. Epilepsia 2001; 42 (03) 316
  • 16 Giulioni M, Acciarri N, Padovani R, Frank F, Galassi E, Gaist G. Surgical management of cavernous angiomas in children. Surg Neurol 1994; 42 (03) 194-199
  • 17 Di Rocco C, Iannelli A, Tamburrini G. Surgical management of paediatric cerebral cavernomas. J Neurosurg Sci 1997; 41 (04) 343-347
  • 18 Xia C, Zhang R, Mao Y, Zhou L. Pediatric cavernous malformation in the central nervous system: report of 66 cases. Pediatr Neurosurg 2009; 45 (02) 105-113
  • 19 Sawarkar DP, Janmatti S, Kumar R. , et al. Cavernous malformations of central nervous system in pediatric patients: our single-centered experience in 50 patients and review of literature. Childs Nerv Syst 2017; 33 (09) 1525-1538
  • 20 Stavrou I, Baumgartner C, Frischer JM, Trattnig S, Knosp E. Long-term seizure control after resection of supratentorial cavernomas: a retrospective single-center study in 53 patients. Neurosurgery 2008; 63 (05) 888-896 , discussion 897
  • 21 Jin Y, Zhao C, Zhang S, Zhang X, Qiu Y, Jiang J. Seizure outcome after surgical resection of supratentorial cavernous malformations plus hemosiderin rim in patients with short duration of epilepsy. Clin Neurol Neurosurg 2014; 119: 59-63
  • 22 Wang C, Yu X, Shrestha S, Qian C, Wang L, Chen G. A predicted model for postoperative seizure outcomes after the surgical resection of supratentorial cavernous malformations. Medicine (Baltimore) 2016; 95 (26) e4078
  • 23 Englot DJ, Han SJ, Lawton MT, Chang EF. Predictors of seizure freedom in the surgical treatment of supratentorial cavernous malformations. J Neurosurg 2011; 115 (06) 1169-1174
  • 24 Yeon JY, Kim JS, Choi SJ, Seo DW, Hong SB, Hong SC. Supratentorial cavernous angiomas presenting with seizures: surgical outcomes in 60 consecutive patients. Seizure 2009; 18 (01) 14-20
  • 25 Hammen T, Romstöck J, Dörfler A, Kerling F, Buchfelder M, Stefan H. Prediction of postoperative outcome with special respect to removal of hemosiderin fringe: a study in patients with cavernous haemangiomas associated with symptomatic epilepsy. Seizure 2007; 16 (03) 248-253
  • 26 Cohen DS, Zubay GP, Goodman RR. Seizure outcome after lesionectomy for cavernous malformations. J Neurosurg 1995; 83 (02) 237-242
  • 27 Cappabianca P, Alfieri A, Maiuri F, Mariniello G, Cirillo S, de Divitiis E. Supratentorial cavernous malformations and epilepsy: seizure outcome after lesionectomy on a series of 35 patients. Clin Neurol Neurosurg 1997; 99 (03) 179-183
  • 28 Moran NF, Fish DR, Kitchen N, Shorvon S, Kendall BE, Stevens JM. Supratentorial cavernous haemangiomas and epilepsy: a review of the literature and case series. J Neurol Neurosurg Psychiatry 1999; 66 (05) 561-568
  • 29 Stefan H, Hammen T. Cavernous haemangiomas, epilepsy and treatment strategies. Acta Neurol Scand 2004; 110 (06) 393-397
  • 30 Baumann CR, Schuknecht B, Lo Russo G. , et al. Seizure outcome after resection of cavernous malformations is better when surrounding hemosiderin-stained brain also is removed. Epilepsia 2006; 47 (03) 563-566
  • 31 Casazza M, Broggi G, Franzini A. , et al. Supratentorial cavernous angiomas and epileptic seizures: preoperative course and postoperative outcome. Neurosurgery 1996; 39 (01) 26-32 , discussion 32–34
  • 32 Fernández S, Miró J, Falip M. , et al. Surgical versus conservative treatment in patients with cerebral cavernomas and nonrefractory epilepsy. Seizure 2012; 21 (10) 785-788
  • 33 Dodrill CB. Problems in the assessment of cognitive effects of antiepileptic drugs. Epilepsia 1992; 33 (Suppl. 06) S29-S32
  • 34 Gallassi R, Morreale A, Lorusso S, Procaccianti G, Lugaresi E, Baruzzi A. Cognitive effects of valproate. Epilepsy Res 1990; 5 (02) 160-164
  • 35 Loring DW, Meador KJ, Lee GP. Determinants of quality of life in epilepsy. Epilepsy Behav 2004; 5 (06) 976-980