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严重隐性营养不良型大疱性表皮松解症患者未受影响皮肤中缺乏VII型胶原蛋白。

Lack of type VII collagen in unaffected skin of patients with severe recessive dystrophic epidermolysis bullosa.

作者信息

Bruckner-Tuderman L, Rüegger S, Odermatt B, Mitsuhashi Y, Schnyder U W

机构信息

Department of Dermatology, University Hospital, Zürich, Switzerland.

出版信息

Dermatologica. 1988;176(2):57-64. doi: 10.1159/000248673.

Abstract

Type VII collagen, the major structural component of the anchoring fibrils, was assayed in normal unaffected skin of patients with different forms of hereditary epidermolysis bullosa. Immunofluorescence staining with affinity-purified polyclonal antibodies to type VII collagen revealed a complete absence of staining in the skin of patients with severe dystrophic recessive epidermolysis bullosa. In all other forms, localized recessive dystrophic, dominant dystrophic, junctional and simplex forms there was an intense continuous linear staining of type VII collagen at the dermoepidermal junction. Also, obligate heterozygote carriers of the gene for severe dystrophic recessive form showed a normal pattern of staining. As internal controls and to define the clinical diagnosis, staining with antibodies to type IV collagen, laminin and bullous pemphigoid antigen was also performed. All these antibodies showed a normal staining pattern indicating an intact general morphology of the dermoepidermal junction zone. These results suggest that there is a defect of type VII collagen in patients with severe recessive dystrophic epidermolysis bullosa. The data also suggest that the group of recessive dystrophic epidermolysis bullosa may be heterogeneous not only clinically, but also at the molecular level.

摘要

VII型胶原蛋白是锚定原纤维的主要结构成分,在患有不同形式遗传性大疱性表皮松解症的患者未受影响的正常皮肤中进行了检测。用针对VII型胶原蛋白的亲和纯化多克隆抗体进行免疫荧光染色显示,严重营养不良性隐性大疱性表皮松解症患者的皮肤中完全没有染色。在所有其他形式中,即局限性隐性营养不良型、显性营养不良型、交界型和单纯型,VII型胶原蛋白在真皮表皮交界处有强烈的连续线性染色。此外,严重营养不良性隐性形式基因的 obligate 杂合子携带者显示出正常的染色模式。作为内部对照并为了明确临床诊断,还用针对IV型胶原蛋白、层粘连蛋白和大疱性类天疱疮抗原的抗体进行了染色。所有这些抗体都显示出正常的染色模式,表明真皮表皮交界区的总体形态完整。这些结果表明,严重隐性营养不良性大疱性表皮松解症患者存在VII型胶原蛋白缺陷。数据还表明,隐性营养不良性大疱性表皮松解症组不仅在临床上可能是异质性的,而且在分子水平上也是如此。

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