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埃勒斯-当洛斯关节过度伸展型(VIIA-B)--扩展表型:从胎儿期到成年期。

Ehlers-Danlos arthrochalasia type (VIIA-B)--expanding the phenotype: from prenatal life through adulthood.

机构信息

Department of Pediatrics, Maastricht University Medical Center, 6202 AZ Maastricht, The Netherlands.

出版信息

Clin Genet. 2012 Aug;82(2):121-30. doi: 10.1111/j.1399-0004.2011.01758.x. Epub 2011 Aug 24.

Abstract

The Ehlers-Danlos syndromes (EDS) form a clinically and genetically heterogeneous group of inherited connective-tissue disorders characterized by joint hypermobility, tissue fragility and skin abnormalities. Six subtypes have been well characterized based on clinical features and molecular genetic abnormalities. The arthrochalasia type EDS (formerly types VIIA and B) is characterized by severe generalized joint hypermobility with multiple dislocations including congenital bilateral dislocation of the hips, muscular hypotonia and distinct dysmorphic features. The diagnosis of the arthrochalasia type EDS is of importance in the neonatal period because of consequences of physical disability in later life. However, the differential diagnosis may be difficult because of overlap with other hypermobility syndromes. In addition, the significant hypotonia may direct the physician toward various neuromuscular diagnoses. As patients become older, the hypotonia decreases and facial features become less distinct. In this report, we describe seven patients at different ages. Timing of diagnosis varied from prenatal life to adult age. The diagnosis of EDS type VII was confirmed by biochemical studies or mutation analysis showing characteristic mutations in COL1A1 and COL1A2. These mutations result in skipping of exon 6, which leads to defective collagen synthesis. For physicians treating patients with EDS type VII, achieving mobility for the patient is the greatest challenge and it may be impossible because of recurrent dislocations of nearly all joints in severe cases.

摘要

埃勒斯-当洛斯综合征(EDS)是一组具有临床和遗传异质性的遗传性结缔组织疾病,其特征为关节过度活动、组织脆弱和皮肤异常。根据临床特征和分子遗传异常,已经很好地描述了六种亚型。关节过度活动型 EDS(以前为 VIIA 和 B 型)的特征为严重的全身性关节过度活动,伴有多处脱位,包括先天性双侧髋关节脱位、肌肉张力减退和明显的畸形特征。由于一生中的身体残疾,关节过度活动型 EDS 的诊断在新生儿期非常重要。然而,由于与其他过度活动综合征的重叠,鉴别诊断可能很困难。此外,显著的张力减退可能会导致医生将其诊断为各种神经肌肉疾病。随着患者年龄的增长,张力减退和面部特征变得不那么明显。在本报告中,我们描述了不同年龄的七名患者。诊断时间从产前到成年不等。EDS 类型 VII 的诊断通过生化研究或突变分析得到证实,这些研究显示 COL1A1 和 COL1A2 中存在特征性突变。这些突变导致外显子 6 的跳跃,从而导致胶原合成缺陷。对于治疗 EDS 类型 VII 患者的医生来说,为患者实现活动能力是最大的挑战,因为在严重情况下,几乎所有关节都会反复脱位,这可能是不可能的。

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