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PMID: 21801164 已发表 · ppublish 英语

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

Clinical genetics ·第 82 卷 ·第 2 期 ·2012-11-13

Klaassens M, Reinstein E, Hilhorst-Hofstee Y, Schrander J J P, Malfait F, Staal H, ten Have L C, Blaauw J, Roggeveen H C J, Krakow D, De Paepe A, van Steensel M A M, Pals G, Graham J M, Schrander-Stumpel C T R M

摘要

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.

文献信息
期刊
Clinical genetics
期刊简称
Clin Genet
发表日期
2012-11-13
收录日期
2012-07-10
更新日期
2016-10-19
语言
英语
国家/地区
Denmark
NLM ID
0253664
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