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脊髓发育不良作为 V.A.C.T.E.R.L 综合征的一个新实体,导致了一个新的缩写 V.A.C.T.E.R.L.S.

Spinal dysraphism as a new entity in V.A.C.TE.R.L syndrome, resulting in a novel acronym V.A.C.TE.R.L.S.

机构信息

Department of Pediatric Neurosurgery, Necker-EM Hospital, Assistance Publique-Hôpitaux de Paris, Université Paris-Descartes, Paris, France.

Department of Visceral Pediatric surgery, Necker-EM Hospital, Assistance Publique-Hôpitaux de Paris, Université Paris-Descartes, Paris, France.

出版信息

Eur J Pediatr. 2020 Jul;179(7):1121-1129. doi: 10.1007/s00431-020-03609-4. Epub 2020 Feb 13.

DOI:10.1007/s00431-020-03609-4
PMID:32055959
Abstract

Anorectal malformation (ARM) is the most common symptom in VACTERL syndrome (vertebral, anal, cardiac, tracheo-esophageal fistula, renal, and limb anomalies). The association of ARM and spinal dysraphisms (DYS) is well documented. We aim to better evaluate children with VACTERL association and ARM, considering the presence or not of DYS. Between 2000 and 2015, 279 children with VACTERL associations were identified in Necker Children's Hospital, Paris. We identified 61 VACTERL children (22%) with ARM. A total of 52 VACTERL children with ARM were included. DYS were identified in 36/52 of cases (69.2%). A total of 33 (63.5%) VACTERL children presented with sphincterial dysfunction. We constated that 28/33 (84.8%) of them had DYS + (p < 0.0001). More children in ARM (DYS +) subgroup are presenting with initial urinary sphincter dysfunction (58 vs 19%, p < 0.009) than ARM (DYS -). We identified 29 lipoma filum in our series, which were not statistically associated with urinary disorders (p = 0.143).Conclusion: We propose to refine the definition of VACTERL association, by adding S as Spinal defect to include it as an integral part of this syndrome, resulting in a novel acronym V.A.C.TE.R.L.S.What is Known:• The VACTERL association: congenital anomalies of the bony vertebral column (V), anorectal malformation (A), congenital cardiopathy (C), tracheo-esophageal defects (TE), renal and urinary tract anomalies (R), and limb malformations (L).• VACTERL children needs a complete appraisal, as early as possible, to adopt the most appropriate therapeutic management.What is New:• Include spine dysraphism (DYS) as a part of this syndrome, resulting in a novel acronym V.A.C.TE.R.L.S.• The significant correlation between VACTERL/DYS and urinary dysfunction requires to investigate the spine cord prenatally.

摘要

肛门直肠畸形(ARM)是 VACTERL 综合征(椎体、肛门、心脏、气管食管瘘、肾脏和肢体异常)中最常见的症状。ARM 与脊柱发育不良(DYS)的关联已有充分的文献记载。我们旨在更好地评估患有 VACTERL 综合征和 ARM 的儿童,考虑是否存在 DYS。2000 年至 2015 年期间,在巴黎 Necker 儿童医院共发现 279 例患有 VACTERL 综合征的儿童。我们发现其中 61 例(22%)患有 ARM。共有 52 例 VACTERL 伴 ARM 的患儿纳入研究。36/52 例(69.2%)患儿存在 DYS。33 例(63.5%)VACTERL 伴 ARM 患儿存在肛门括约肌功能障碍。我们发现,33 例中有 28 例(84.8%)患儿存在 DYS(p<0.0001)。ARM(DYS+)亚组中,更多的患儿(58%)初始存在尿失禁(58%),而 ARM(DYS-)亚组中为 19%(p<0.009)。我们的研究中发现 29 例脊髓脊膜膨出,但与尿失禁无统计学相关性(p=0.143)。结论:我们建议通过添加 S 作为脊柱缺陷,将其纳入 VACTERL 综合征的定义中,以将其作为该综合征的一个组成部分,从而提出一个新的缩写词 V.A.C.TE.R.L.S。已知情况:• VACTERL 综合征:先天性椎体骨骼异常(V)、肛门直肠畸形(A)、先天性心脏病(C)、气管食管缺陷(TE)、肾脏和泌尿道异常(R)以及肢体畸形(L)。• VACTERL 患儿需要尽早进行全面评估,以便采取最合适的治疗管理。新发现:• 将脊柱发育不良(DYS)纳入该综合征,提出一个新的缩写词 V.A.C.TE.R.L.S。• VACTERL/DYS 与尿失禁之间存在显著相关性,这需要在产前对脊髓进行检查。

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本文引用的文献

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The etiology of VACTERL association: Current knowledge and hypotheses.VACTERL 协会的病因:现有知识和假说。
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Finding patients using similarity measures in a rare diseases-oriented clinical data warehouse: Dr. Warehouse and the needle in the needle stack.在面向罕见病的临床数据仓库中使用相似性度量来寻找患者:仓库博士与针堆中的针。
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肛门直肠畸形合并脊髓异常。
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The genetic landscape and clinical implications of vertebral anomalies in VACTERL association.VACTERL综合征中椎体异常的遗传图谱及临床意义
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Neural tube dysraphism: review of cutaneous markers and imaging.神经管闭合不全:皮肤标志物与影像学综述
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Impact of spinal dysraphism on urinary and faecal prognosis in 25 cases of cloacal malformation.25 例泄殖腔畸形患者的脊柱发育不良对尿便预后的影响。
J Pediatr Urol. 2014 Dec;10(6):1199-205. doi: 10.1016/j.jpurol.2014.05.012. Epub 2014 Jul 19.
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Clinical and etiological heterogeneity in patients with tracheo-esophageal malformations and associated anomalies.气管食管畸形及相关异常患者的临床和病因学异质性。
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Pediatr Surg Int. 2012 Jul;28(7):725-9. doi: 10.1007/s00383-012-3073-y. Epub 2012 Mar 16.