Early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report

Abstract Background Infantile spasms (IS) are the most common childhood epileptic encephalopathy. Focal cortical dysplasia (FCD) and gray matter heterotopias (GH) are common structural causes of IS. The recommended first-line treatment for IS patients with structural causes is surgical intervention,...

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Main Authors: Haiyan Yang, Zhiquan Yang, Jing Peng, Yehong Huang, Zhuanyi Yang, Fei Yin, Liwen Wu
Format: Article
Language:English
Published: BMC 2020-10-01
Series:Acta Epileptologica
Subjects:
Online Access:http://link.springer.com/article/10.1186/s42494-020-00025-x
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spelling doaj-2133e6385355442783055804668015212020-11-25T01:19:18ZengBMCActa Epileptologica2524-44342020-10-01211610.1186/s42494-020-00025-xEarly surgical intervention for structural infantile spasms in two patients under 6 months old: a case reportHaiyan Yang0Zhiquan Yang1Jing Peng2Yehong Huang3Zhuanyi Yang4Fei Yin5Liwen Wu6Department of Pediatrics, Xiangya Hospital, Central South UniversityDepartment of Neurosurgery, Xiangya Hospital, Central South UniversityDepartment of Pediatrics, Xiangya Hospital, Central South UniversityDepartment of Pediatrics, Xiangya Hospital, Central South UniversityDepartment of Neurosurgery, Xiangya Hospital, Central South UniversityDepartment of Pediatrics, Xiangya Hospital, Central South UniversityDepartment of Neurology, Hunan Children’s HospitalAbstract Background Infantile spasms (IS) are the most common childhood epileptic encephalopathy. Focal cortical dysplasia (FCD) and gray matter heterotopias (GH) are common structural causes of IS. The recommended first-line treatment for IS patients with structural causes is surgical intervention, according to the International League Against Epilepsy (ILAE) commission guidelines. However, there is currently no consensus on appropriate timings of surgery. Case presentations Two structural IS cases are presented here: one was caused by FCD, and the other by GH. Both patients exhibited recurrent seizures at the age of 2 months, had poor responses to various antiepileptic drugs (AEDs) and displayed severe mental and motor developmental retardation. Seizure types included focal seizures and spasms. Brain magnetic resonance imaging showed abnormal gray signal or suspicious FCD lesions that coincided with the origin of the focal seizures. The patients underwent lesion resection before the age of 6 months. Follow-up observation showed that seizures of both patients were completely controlled several days after the surgery. All AEDs were gradually reduced in dosage within 1 year, and the mental and motor development almost returned to normal. Conclusion Early resection of lesions in structural IS patients has benefits of effectively controlling convulsions and improving developmental retardation. Infants at several months of age can well tolerate craniotomy, and their cognitive development is more likely to return to normal after early surgery.http://link.springer.com/article/10.1186/s42494-020-00025-xInfantile spasmsFocal cortical dysplasiaGray matter heterotopiasEarly lesion resection
collection DOAJ
language English
format Article
sources DOAJ
author Haiyan Yang
Zhiquan Yang
Jing Peng
Yehong Huang
Zhuanyi Yang
Fei Yin
Liwen Wu
spellingShingle Haiyan Yang
Zhiquan Yang
Jing Peng
Yehong Huang
Zhuanyi Yang
Fei Yin
Liwen Wu
Early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report
Acta Epileptologica
Infantile spasms
Focal cortical dysplasia
Gray matter heterotopias
Early lesion resection
author_facet Haiyan Yang
Zhiquan Yang
Jing Peng
Yehong Huang
Zhuanyi Yang
Fei Yin
Liwen Wu
author_sort Haiyan Yang
title Early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report
title_short Early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report
title_full Early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report
title_fullStr Early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report
title_full_unstemmed Early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report
title_sort early surgical intervention for structural infantile spasms in two patients under 6 months old: a case report
publisher BMC
series Acta Epileptologica
issn 2524-4434
publishDate 2020-10-01
description Abstract Background Infantile spasms (IS) are the most common childhood epileptic encephalopathy. Focal cortical dysplasia (FCD) and gray matter heterotopias (GH) are common structural causes of IS. The recommended first-line treatment for IS patients with structural causes is surgical intervention, according to the International League Against Epilepsy (ILAE) commission guidelines. However, there is currently no consensus on appropriate timings of surgery. Case presentations Two structural IS cases are presented here: one was caused by FCD, and the other by GH. Both patients exhibited recurrent seizures at the age of 2 months, had poor responses to various antiepileptic drugs (AEDs) and displayed severe mental and motor developmental retardation. Seizure types included focal seizures and spasms. Brain magnetic resonance imaging showed abnormal gray signal or suspicious FCD lesions that coincided with the origin of the focal seizures. The patients underwent lesion resection before the age of 6 months. Follow-up observation showed that seizures of both patients were completely controlled several days after the surgery. All AEDs were gradually reduced in dosage within 1 year, and the mental and motor development almost returned to normal. Conclusion Early resection of lesions in structural IS patients has benefits of effectively controlling convulsions and improving developmental retardation. Infants at several months of age can well tolerate craniotomy, and their cognitive development is more likely to return to normal after early surgery.
topic Infantile spasms
Focal cortical dysplasia
Gray matter heterotopias
Early lesion resection
url http://link.springer.com/article/10.1186/s42494-020-00025-x
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