Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a Child

Linear scleroderma is a form of localized scleroderma characterized by sclerotic lesions distributed in a linear, band-like pattern. The “en coup de sabre” subtype of linear scleroderma is more often associated with systemic morbidity, including ocular, oral, and neurological abnormalities. Here, we...

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Main Authors: Kuo-Liang Chiang, Kai-Ping Chang, Tai-Tong Wong, Ting-Rong Hsu
Format: Article
Language:English
Published: Elsevier 2009-12-01
Series:Pediatrics and Neonatology
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Online Access:http://www.sciencedirect.com/science/article/pii/S1875957209600814
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author Kuo-Liang Chiang
Kai-Ping Chang
Tai-Tong Wong
Ting-Rong Hsu
author_facet Kuo-Liang Chiang
Kai-Ping Chang
Tai-Tong Wong
Ting-Rong Hsu
author_sort Kuo-Liang Chiang
collection DOAJ
description Linear scleroderma is a form of localized scleroderma characterized by sclerotic lesions distributed in a linear, band-like pattern. The “en coup de sabre” subtype of linear scleroderma is more often associated with systemic morbidity, including ocular, oral, and neurological abnormalities. Here, we report one patient with typical linear scleroderma “en coup de sabre” (LSCS). Initially, he presented with refractory partial seizures before the characteristic skins lesion on his head developed. This was a rare case with obvious brain parenchyma involvement. We did not prescribe medication but performed serial brain magnetic resonance imaging follow-up for the intraparenchymal lesion. The atrophic changes of the skin, face and brain remained the same, and his seizures had not worsened at the most recent follow-up. Parry-Romberg syndrome, a very similar condition, should be differentiated from LSCS.
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publisher Elsevier
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series Pediatrics and Neonatology
spelling doaj-art-431e1883b65e458282efb9b9e5dddafd2025-08-20T01:56:56ZengElsevierPediatrics and Neonatology1875-95722009-12-0150629429810.1016/S1875-9572(09)60081-4Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a ChildKuo-Liang Chiang0Kai-Ping Chang1Tai-Tong Wong2Ting-Rong Hsu3Department of Pediatrics, Neurological Institute, Taipei Veterans General Hospital, Taipei, TaiwanDepartment of Pediatrics, Neurological Institute, Taipei Veterans General Hospital, Taipei, TaiwanDepartment of Pediatrics, Kuang-Tien General Hospital, Taichung, TaiwanDepartment of Pediatrics, Neurological Institute, Taipei Veterans General Hospital, Taipei, TaiwanLinear scleroderma is a form of localized scleroderma characterized by sclerotic lesions distributed in a linear, band-like pattern. The “en coup de sabre” subtype of linear scleroderma is more often associated with systemic morbidity, including ocular, oral, and neurological abnormalities. Here, we report one patient with typical linear scleroderma “en coup de sabre” (LSCS). Initially, he presented with refractory partial seizures before the characteristic skins lesion on his head developed. This was a rare case with obvious brain parenchyma involvement. We did not prescribe medication but performed serial brain magnetic resonance imaging follow-up for the intraparenchymal lesion. The atrophic changes of the skin, face and brain remained the same, and his seizures had not worsened at the most recent follow-up. Parry-Romberg syndrome, a very similar condition, should be differentiated from LSCS.http://www.sciencedirect.com/science/article/pii/S1875957209600814intractable seizurelinear scleroderma en coup de sabreprogressive brain atrophy
spellingShingle Kuo-Liang Chiang
Kai-Ping Chang
Tai-Tong Wong
Ting-Rong Hsu
Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a Child
Pediatrics and Neonatology
intractable seizure
linear scleroderma en coup de sabre
progressive brain atrophy
title Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a Child
title_full Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a Child
title_fullStr Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a Child
title_full_unstemmed Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a Child
title_short Linear Scleroderma “En Coup De Sabre”: Initial Presentation as Intractable Partial Seizures in a Child
title_sort linear scleroderma en coup de sabre initial presentation as intractable partial seizures in a child
topic intractable seizure
linear scleroderma en coup de sabre
progressive brain atrophy
url http://www.sciencedirect.com/science/article/pii/S1875957209600814
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AT kaipingchang linearsclerodermaencoupdesabreinitialpresentationasintractablepartialseizuresinachild
AT taitongwong linearsclerodermaencoupdesabreinitialpresentationasintractablepartialseizuresinachild
AT tingronghsu linearsclerodermaencoupdesabreinitialpresentationasintractablepartialseizuresinachild