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Postoperative Incidence of Seizure and Cerebral Infarction in Pediatric

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Postoperative Incidence of Seizure and Cerebral Infarction in Pediatric Liu et al. Chinese Neurosurgical Journal (2021) 7:11 https://doi.org/10.1186/s41016-020-00224-y 中华医学会神经外科学分会 CHINESE MEDICAL ASSOCIATION CHINESE NEUROSURGICAL SOCIETY RESEARCH Open Access Postoperative incidence of seizure and cerebral infarction in pediatric patients with epileptic type moyamoya disease: a meta- analysis of single rate Jingjing Liu1,2, Qinlan Xu1, Hongchuan Niu3, Rong Wang3,4, Xun Ye3,4* and Xianzeng Liu1* Abstract Background: Surgery is a conventional mature treatment for moyamoya disease (MMD). However, whether surgery is also an effective therapy for epileptic type MMD has seldom been investigated systematically. The study aims to summarize the pooled postoperative incidence of seizure and cerebral infarction in pediatric patients with epileptic type moyamoya disease. Method: The study was a systematic review and critical appraisal with a meta-analysis of cohort studies, both prospective and retrospective. Studies were identified by a computerized search of PubMed, Embase, Web of Science, Wanfang, and CNKI databases. In a literature search, a total of 7 cohort studies were identified. The I2statistic was used to quantify heterogeneity. A fixed-effect model was used to synthesize the results. The linear regression test of funnel plot asymmetry was used to estimate the potential publication bias. Results: The pooled estimated postoperative incidence of seizure in pediatric patients with epileptic type moyamoya disease was 23.44%. The pooled estimated postoperative incidence of cerebral infarction in pediatric patients with epileptic type moyamoya disease was 9.12%. Low substantial heterogeneity and potential publication bias were present. Conclusions: Evidence from this study suggests that the postoperative incidence of seizure and cerebral infarction is relatively low. Surgery is an effective and secure therapy for pediatric patients with epileptic type moyamoya disease. Keywords: Moyamoya disease, Epileptic type, Seizure, Ischemic event, Surgery * Correspondence: [email protected]; [email protected] Jingjing Liu is the first and responsible author. Xianzeng Liu and Xun Ye are the equal corresponding authors. 3Peking University International Hospital, Department of Neurosurgery, Peking University, Beijing, China 1Peking University International Hospital, Department of Neurology, Peking University, Beijing, China Full list of author information is available at the end of the article © The Author(s). 2021 Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. Liu et al. Chinese Neurosurgical Journal (2021) 7:11 Page 2 of 8 Background Some of the epileptic type patients suffer seizures as Moyamoya disease (MMD) is a type of progressive oc- the first symptom, and others may be secondary to is- clusive cerebrovascular disease, with the significant char- chemic stroke [6]. The clinical manifestation and pro- acteristic of steno or blocked blood vessels at the end of gression of this type may be non-specific [5]. Some the internal carotid artery (ICA), proximal middle cere- scholars believe that epileptic seizure in epileptic type bral artery (MCA), and anterior cerebral artery (ACA) patients is not a specific symptom but mostly caused by [1]. The name “moyamoya” means “puff of smoke” in cerebral ischemia [7]. The cause of epilepsy in MMD Japanese and describes the appearance of the formation could be various, including ischemic or hemorrhagic of smoke-like abnormal blood vessels in the base of the stroke [5], hyperperfusion [8], and so on. About 20–30% skull in cerebral angiography. It frequently occurs in the of MMD patients present with seizures [9], but only 3– East Asian population, may cause ischemic or 4% epileptic type MMD without vascular events [10, 11]. hemorrhage stroke, epilepsy, headache, or transient is- Revascularization operation is effective for MMD pre- chemic attack (TIA) [2]. The epileptic seizure is the sec- senting with the cerebral ischemic event has been ond common symptom of MMD in pediatric patients proved by multiple studies [11–14]. Surgery could re- [3], and the third most common manifestation of MMD duce the frequency of TIAs and cerebral infarction, and in all patients [4].MMD whose main clinical symptoms improve the long-term prognosis of brain functions. The are convulsive seizures is called “epileptic type moya- cerebral hemodynamics and metabolism are also im- moya disease” [5]. The treatment methods and clinical proved following surgery treatment, which could be eval- outcomes for this type of MMD are seldom reported. uated by SPECT or PET [13]. Both direct and indirect Fig. 1 Flow chart of the literature searches for the systematic review Liu et al. Chinese Neurosurgical Journal (2021) 7:11 Page 3 of 8 revascularizations or the combination of these two types postoperative seizure cases, and (11) the number of post- could obtain satisfactory results. Since epileptic type, operative cerebral infarction cases. MMD has rarely been discussed in detail before, whether surgery could improve these patients still lack research Statistical analysis systematically. In this study, we will summarize the Meta-analyses of a single rate were carried out by using pooled postoperative incidence of seizure and cerebral RStudio version 3.6.1 (2019-07-05). The I2 statistic was infarction in pediatric patients with epileptic type MMD, used to quantify heterogeneity (I2 < 25%, low heterogen- in order to estimate the effect of the surgery treatment eity). The fixed-effects models were used to calculate an on these patients. overall pooled proportion and 95% CI when the results presented as low heterogeneity. Linear regression test of Methods funnel plot asymmetry was used to estimate the poten- Literature search tial publication bias (P > 0.05, no publication bias). Influ- We computerized searched the PubMed, Web of Sci- ential analysis was used to detect whether there were ence, Embase, Wanfang, and CNKI databases. A com- some studies exert a very high influence on our overall bination of keywords and similar strategies was used to results. identify previously published studies. The medical sub- ject heading (MeSH) key words were moyamoya disease, Results epilepsy or seizure, and surgery or operation. Two inde- All of the details of the included studies are listed in pendent researchers (Liu J, Xu X) conducted separate lit- Table 1. erature searches. The last update for research was done The summarized pooled postoperative incidence rate in August 2019. No limitation on language was consid- of seizure in pediatric patients with epileptic type MMD ered. After the removal of duplicate articles, a total of in the seven studies using the fixed-effects models was 523 articles were found (Fig. 1). 23.44% (95% CI 14.39–33.63%) with low heterogeneity (Tau2 =0,I2 = 0% (95%CI 0–67.2%), P = 0.5) (Fig. 2 a). Criteria for inclusion and exclusion Low publication bias was estimated by the linear regres- Articles were included in the meta-analysis if they met sion test of funnel plot asymmetry (P = 0.33) (Fig. 3a). the following criteria: (1) prospective and retrospective The summarized pooled postoperative incidence rate cohort studies on patients with epileptic type MMD, (2) of seizure in pediatric patients with epileptic type MMD enrolled patients are younger than 18 years old, (3) at would change ≤ 2.6% after the exclusion of any studies. least 5 epileptic type MMD patients were included in The maximum increase of the rate would be 2.6% after the study, (4) chose direct and indirect revascularizations the exclusion of the study by Nakase H, et al. (1993) [6], or the combination surgery treatment for these patients, whereas the maximum reduction would be 1.9% after (5) the prognosis of these patients after surgery was de- the exclusion of the study by Ulrich PT, et al. (2011) scribed in detail, including the postoperative incidence [16]. Removing any of the findings of these studies will of seizure and cerebral infarction. We excluded all of the not make the heterogeneity of the studies significant (I2 reviews, case reports, articles without full text, or not be = 0%). translated into English or Chinese. We also excluded the The summarized pooled postoperative incidence rate studies without detail about epileptic type MMD, surgi- of cerebral infarction in pediatric patients with epileptic cal treatment, or outcome analysis after surgery. No ran- type MMD in the four studies using the fixed-effects domized controlled trials were found. Finally, seven models was 9.12% (95% CI 0.75–22.34%) with low het- cohorts
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