RESEARCH PAPER
Epilepsy in paediatric patients with schizencephaly
| 1 | Department of Paediatric Neurology, School of Medicine, Medical University of Silesia, Katowice, Poland |
| 2 | Department of Biostatistics, School of Public Health in Bytom, Medical University of Silesia, Katowice, Poland |
| 3 | Institute of Statistics and Demography, School of Economic Warsaw, Poland |
| 4 | Department of Women’s Health, Institute of Rural Health, Lublin, Poland |
| 5 | Department of Basic Biomedical Science, School of Pharmacy with the Division of Laboratory Medicine in Sosnowiec, Medical University of Silesia, Katowice, Poland |
CORRESPONDING AUTHOR
Beata Sarecka-Hujar
Department of Basic Biomedical Science, School of Pharmacy with the Division of Laboratory Medicine in Sosnowiec, Medical University of Silesia in Katowice
Department of Basic Biomedical Science, School of Pharmacy with the Division of Laboratory Medicine in Sosnowiec, Medical University of Silesia in Katowice
Ann Agric Environ Med. 2020;27(2):279–283
KEYWORDS
TOPICS
ABSTRACT
Introduction:
Schizencephaly is one of the rare congenital defects of the central nervous system (CNS), known as neuronal migration disorders. The etiology of schizencecephaly is unequivocal. Established etiologies include in-utero infections (cytomegalovirus and herpes simplex virus, HSV type I), toxic abuse (cocaine, alcohol), as well as drug use (warfarin).
Objectives:
he aim of the study was to analyze the clinical presentation of schizencephaly with particular consideration of the course of epilepsy in paediatric patients.
Material and methods:
The study group consisted of 38 children with schizencephaly (20 of them had seizure) and was retrospectively assessed. Data were analyzed using SAS version 9.4. U Mann-Whitney and χ 2 tests and logistic regression analysis were used in statistical analyses.
Results:
Epilepsy was the most frequent in bilateral type II schizencephaly (p=0.033). In logistic regression analysis, the presence of bilateral open schizencephaly significantly increased the risk of seizures (OR=11.67; 95%CI 2.44–55.83; p=0.002). Drug-resistant epilepsy was observed in 9 children (45% of the children with epilepsy). Prevalence of both epilepsy and drug-resistant epilepsy in schizencephaly did not significantly depend on gender, stage of development, type or localization of schizencephaly, and other coexisting CNS defects or clinical presentation of schizencephaly at follow-up in the study group of patients.
Conclusions:
The bilateral type of schizencephaly was identified as an independent risk factor for epilepsy in the analyzed children.
Schizencephaly is one of the rare congenital defects of the central nervous system (CNS), known as neuronal migration disorders. The etiology of schizencecephaly is unequivocal. Established etiologies include in-utero infections (cytomegalovirus and herpes simplex virus, HSV type I), toxic abuse (cocaine, alcohol), as well as drug use (warfarin).
Objectives:
he aim of the study was to analyze the clinical presentation of schizencephaly with particular consideration of the course of epilepsy in paediatric patients.
Material and methods:
The study group consisted of 38 children with schizencephaly (20 of them had seizure) and was retrospectively assessed. Data were analyzed using SAS version 9.4. U Mann-Whitney and χ 2 tests and logistic regression analysis were used in statistical analyses.
Results:
Epilepsy was the most frequent in bilateral type II schizencephaly (p=0.033). In logistic regression analysis, the presence of bilateral open schizencephaly significantly increased the risk of seizures (OR=11.67; 95%CI 2.44–55.83; p=0.002). Drug-resistant epilepsy was observed in 9 children (45% of the children with epilepsy). Prevalence of both epilepsy and drug-resistant epilepsy in schizencephaly did not significantly depend on gender, stage of development, type or localization of schizencephaly, and other coexisting CNS defects or clinical presentation of schizencephaly at follow-up in the study group of patients.
Conclusions:
The bilateral type of schizencephaly was identified as an independent risk factor for epilepsy in the analyzed children.
REFERENCES (26)
1.
Howe DT, Rankin J, Draper E. Schizencephaly prevalence, prenatal diagnosis and clues to etiology: a register-based study. Ultrasound Obstet Gynecol. 2012; 39: 75–82.
2.
Granata T, Battaglia G. Schizencephaly. Handb Clin Neurol. 2008; 87: 235–246.
3.
Szabo N, Gyurgyinka G, Kobor J, Bereg E, Turi S, Sztriha L. Epidemiology and clinical spectrum of schizencephaly in south-eastern Hungary. J Child Neurol. 2010; 25(11): 1335–1339.
4.
Miller GM, Stears JC, Guggenheim MA, Wilkening GN. Schizencephaly: a clinical and CT study. Neurology. 1984; 34: 997–1001.
5.
Leventer RJ, Phelan EM, Coleman LT, Kean MJ, Jackson GD, Harvey AS. Clinical and imaging features of cortical malformations in childhood. Neurology. 1999; 53: 715–722.
6.
Verrotti A, Spalice A, Ursitti F, Papetti L, Mariani R, Castronovo A, Mastrangelo M, Iannetti P. New trends in neuronal migration disorders. Eur J Paediatr Neurol. 2010; 14(1): 1–12.
7.
Curry CJ, Lammer EJ, Nelson V, Shaw GM. Schizencephaly: Heterogenous etiologies in a population of 4 million California births. Am J Med Genet. 2005; 137A: 181–189.
8.
Dies KA, Bodell A, Hisama FM, Guo CY, Barry B, Chang BS, Barkovich AJ, Walsh CA. Schizencephaly: association with young maternal age, alcohol abuse and lack of prenatal care. J Child Neurol. 2013; 28(2): 198–203.
9.
Denis D, Chateil JF, Brun M, Brissaud O, Lacombe D, Fontan D, Flurin V, Pedespan J. Schizencephaly: clinical and imaging features in 30 infantile cases. Brain Dev. 2000; 22: 475–483.
10.
Granata T, Freri E, Caccia C, Setola V, Taroni F, Battaglia G. Schizencephaly: clinical spectrum, epilepsy and pathogenesis. J Child Neurol. 2005; 20: 313–318.
11.
Berg AT, Berkovic SF, Brodie MJ, Buchhalter J, Cross JH, van Emde Boas W, Engel J, French J, Glauser TA, Mathern GW, Moshé SL, Nordli D, Plouin P, Scheffer IE. Revised terminology and concepts for organization of seizures and epilepsies: Report of the ILAE Commission on Classification and Terminology,2005–2009. Epilepsia 2010; 51(4): 676–685.
12.
Fisher RS, van Emde Boas W, Blume W, Elger C, Genton P, Lee P, Engel J Jr. Epileptic seizures and epilepsy: definitions proposed by the International League Against Epilepsy (ILAE) and the International Bureau for Epilepsy (IBE). Epilepsia. 2005; 46(4): 470–472.
13.
Kwan P, Arzimanoglou A, Berg AT, Brodie MJ, Allen Hauser W, Mathern G, Moshé SL, Perucca E, Wiebe S, French J. Definition of drug resistant epilepsy: consensus proposal by the ad hoc Task Force of the ILAE Commission on Therapeutic Strategies. Epilepsia. 2010; 51(6): 1069–1077.
14.
Hung P-C, Wang H-S, Chou M-L, Lin K-L, Hsieh M-Y, Chou I-J, Wong A M-C. Schizencephaly in children: A single medical center retrospective study. Pediatrics and Neonatology 2018; 59: 573–580.
15.
Stopa J, Kucharska-Miąsik I, Dziurzyńska-Białek E, Kostkiewicz A, Solińska A, Zając-Mnich M, Guz W, Samojedny A. Diagnosting imaging and problems of schizencephaly. Pol J Radiol. 2014; 79: 444–449.
16.
Kułak W, Okurowska-Zawada B, Gościk E, Sienkiewicz D, Paszko-Patej G, Kubas B. Schizencephaly as a cause of spastic cerebral palsy. Adv Med Sci. 2011; 56(1): 64–70.
17.
Packard AM, Miller VS, Delgado MR. Schizencephaly: correlations of clinical and radiological features. Neurology 1997; 48(5): 1427–1434.
18.
Pascual-Castroviejo I, Pascual-Pascual SI, Velazquez-Fragua R, Viaño J, Quiñones D. Schizencephaly: a study of 16 patients. Neurologia. 2012; 27(8): 491–499.
19.
Sarecka-Hujar B, Kopyta I, Raczkiewicz D. Risk factors, types and outcomes of arterial ischemic stroke in Polish pediatric patients: A retrospective single-center study. Arch Med Sci 2018, DOI: https://doi.org/10.5114/aoms.2....
20.
Chmielewska B, Lis K, Rejdak K, Balcerzak M, Steinborn B. Pattern of adverse events of antiepileptic drugs: results of the aESCAPE study in Poland. Arch Med Sci. 2013; 9(5): 858–864.
21.
Lopes CF, Cendes F, Piovesana AM, Torres F, Lopes-Cendes I, Montenegro MA, Guerreiro MM. Epileptic features of patients with unilateral and bilateral schizencephaly. J Child Neurol. 2006; 21(9): 757–760.
22.
Hayashi N, Tsutsumi Y, Barkovich AJ. Morphological features and associated anomalies of schizencephaly in the clinical population. Neuroradiology. 2002; 44(5): 418–427.
23.
Okunlola AI, Olowoyo P, Okunlola CK, Babalola OF. Adult-Onset Seizure Disorder Secondary to Schizencephaly. Asian J Neurosurg. 2020; 15(1): 159–161.
24.
Cusmai R, Verrotti A, Moavero R, Curatolo P, Battaglia D, Matricardi S, Spalice A, Vigevano F, Pruna D, Parisi P, D’Aniello A, Di Gennaro G, Coppola G. Rufinamide for the treatment of refractory epilepsy secondary to neuronal migration disorders. Epilepsy Res. 2014; 108(3): 542–546.
25.
Verrotti A, Loiacono G, Rossi A, Tartaro A, Delli Pizzi A, Coppola G. Successful Treatment of Refractory Seizures With Rufinamide in Children With Schizencephaly: Report of 3 Cases. J Child Neurol. 2015; 30(8): 1079–1083.
26.
Kanner AM, Ashman E, Gloss D, Harden C, Bourgeois B, Bautista JF, Abou-Khalil B, Burakgazi-Dalkilic E, Park EL, Stern J, Hirtz D, Nespeca M, Gidal B, Faught E, French J. Practice guideline update summary: Efficacy and tolerability of the new antiepileptic drugs II: Treatment-resistant epilepsy: Report of the American Epilepsy Society and the Guideline Development, Dissemination, and Implementation Subcommittee of the American Academy of Neurology. Epilepsy Curr. 2018; 18(4): 269–278.
Improvement of visual identification of the journal - task financed under the agreement No. 618/P-DUN/2019 by the Minister of Science and Higher Education allocated to the activities of disseminating science.
Generation of the DOI (Digital Object Identifier) - task financed under the agreement No. 618/P-DUN/2019 by the Minister of Science and Higher
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