杜氏和贝克尔肌营养不良症的癫痫特征。

Praveen Kumar Ramani, Kindann Fawcett, Debra Guntrum, Hallie Samuel, Emma Ciafaloni, Aravindhan Veerapandiyan
{"title":"杜氏和贝克尔肌营养不良症的癫痫特征。","authors":"Praveen Kumar Ramani,&nbsp;Kindann Fawcett,&nbsp;Debra Guntrum,&nbsp;Hallie Samuel,&nbsp;Emma Ciafaloni,&nbsp;Aravindhan Veerapandiyan","doi":"10.1177/2329048X231159484","DOIUrl":null,"url":null,"abstract":"<p><p>Dystrophinopathies cover a spectrum of X-linked muscle disorders including Duchenne muscular dystrophy (DMD), Becker muscular dystrophy (BMD), and cardiomyopathy due to pathogenic variants in the <i>DMD</i> gene. Neuropsychiatric manifestations occur approximately in one-third of patients with dystrophinopathy. Epilepsy has been described. Here we report seizure and electroencephalographic features of boys with dystrophinopathy and epilepsy. This is a retrospective chart review of eight patients with dystrophinopathy and epilepsy seen at Arkansas Children's Hospital and University of Rochester Medical center. Six patients had DMD and two had BMD. Five patients had generalized epilepsy. Three patients had focal epilepsy and the seizures were intractable in two of them. Brain imaging was available for five patients and were within normal limits. EEG abnormalities were noted in six patients. Seizures were well controlled on the current antiepileptic medication regimen in all patients. Further research is needed to better elucidate the underlying mechanisms and genotype-phenotype correlations.</p>","PeriodicalId":72572,"journal":{"name":"Child neurology open","volume":"10 ","pages":"2329048X231159484"},"PeriodicalIF":0.0000,"publicationDate":"2023-01-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/22/95/10.1177_2329048X231159484.PMC9950600.pdf","citationCount":"0","resultStr":"{\"title\":\"Epilepsy Characteristics in Duchenne and Becker Muscular Dystrophies.\",\"authors\":\"Praveen Kumar Ramani,&nbsp;Kindann Fawcett,&nbsp;Debra Guntrum,&nbsp;Hallie Samuel,&nbsp;Emma Ciafaloni,&nbsp;Aravindhan Veerapandiyan\",\"doi\":\"10.1177/2329048X231159484\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Dystrophinopathies cover a spectrum of X-linked muscle disorders including Duchenne muscular dystrophy (DMD), Becker muscular dystrophy (BMD), and cardiomyopathy due to pathogenic variants in the <i>DMD</i> gene. Neuropsychiatric manifestations occur approximately in one-third of patients with dystrophinopathy. Epilepsy has been described. Here we report seizure and electroencephalographic features of boys with dystrophinopathy and epilepsy. This is a retrospective chart review of eight patients with dystrophinopathy and epilepsy seen at Arkansas Children's Hospital and University of Rochester Medical center. Six patients had DMD and two had BMD. Five patients had generalized epilepsy. Three patients had focal epilepsy and the seizures were intractable in two of them. Brain imaging was available for five patients and were within normal limits. EEG abnormalities were noted in six patients. Seizures were well controlled on the current antiepileptic medication regimen in all patients. Further research is needed to better elucidate the underlying mechanisms and genotype-phenotype correlations.</p>\",\"PeriodicalId\":72572,\"journal\":{\"name\":\"Child neurology open\",\"volume\":\"10 \",\"pages\":\"2329048X231159484\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2023-01-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://ftp.ncbi.nlm.nih.gov/pub/pmc/oa_pdf/22/95/10.1177_2329048X231159484.PMC9950600.pdf\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Child neurology open\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1177/2329048X231159484\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Child neurology open","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1177/2329048X231159484","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0

摘要

肌营养不良症涵盖了一系列x连锁肌肉疾病,包括杜氏肌营养不良症(DMD)、贝克肌营养不良症(BMD)和由DMD基因致病性变异引起的心肌病。大约三分之一的肌营养不良患者出现神经精神症状。癫痫已经被描述过了。在此,我们报告患有肌营养不良症和癫痫的男孩的癫痫发作和脑电图特征。这是对阿肯色儿童医院和罗切斯特大学医学中心的8例肌营养不良症和癫痫患者的回顾性分析。6例为DMD, 2例为BMD。5例患者有全身性癫痫。3例患者有局灶性癫痫,其中2例癫痫发作难治性。5例患者的脑成像在正常范围内。6例患者脑电图异常。在目前的抗癫痫药物治疗方案下,所有患者的癫痫发作都得到了很好的控制。需要进一步的研究来更好地阐明潜在的机制和基因型-表型相关性。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

摘要图片

摘要图片

查看原文
分享 分享
微信好友 朋友圈 QQ好友 复制链接
本刊更多论文
Epilepsy Characteristics in Duchenne and Becker Muscular Dystrophies.

Dystrophinopathies cover a spectrum of X-linked muscle disorders including Duchenne muscular dystrophy (DMD), Becker muscular dystrophy (BMD), and cardiomyopathy due to pathogenic variants in the DMD gene. Neuropsychiatric manifestations occur approximately in one-third of patients with dystrophinopathy. Epilepsy has been described. Here we report seizure and electroencephalographic features of boys with dystrophinopathy and epilepsy. This is a retrospective chart review of eight patients with dystrophinopathy and epilepsy seen at Arkansas Children's Hospital and University of Rochester Medical center. Six patients had DMD and two had BMD. Five patients had generalized epilepsy. Three patients had focal epilepsy and the seizures were intractable in two of them. Brain imaging was available for five patients and were within normal limits. EEG abnormalities were noted in six patients. Seizures were well controlled on the current antiepileptic medication regimen in all patients. Further research is needed to better elucidate the underlying mechanisms and genotype-phenotype correlations.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
自引率
0.00%
发文量
0
审稿时长
15 weeks
期刊最新文献
Corrigendum to "Refractory Jeavons Syndrome from Birth Symptomatic to PLCB1 Mutation". Recurrent Spinal Arteriovenous Malformations in a Patient with Cobb Syndrome. “Oh My Sleeping Child” … Narcolepsy Type 1 in a 22-Month-Old Boy Cortical Hand Knob Paradoxical Thromboembolic Stroke in an Adolescent with Secundum Atrial Septal Defect and Paget-Schroetter Syndrome A Severe Case of Streptococcus pneumoniae Meningoencephalitis in an Infant Resulting in Fatal Strokes
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
现在去查看 取消
×
提示
确定
0
微信
客服QQ
Book学术公众号 扫码关注我们
反馈
×
意见反馈
请填写您的意见或建议
请填写您的手机或邮箱
已复制链接
已复制链接
快去分享给好友吧!
我知道了
×
扫码分享
扫码分享
Book学术官方微信
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术
文献互助 智能选刊 最新文献 互助须知 联系我们:info@booksci.cn
Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。
Copyright © 2023 Book学术 All rights reserved.
ghs 京公网安备 11010802042870号 京ICP备2023020795号-1