Wong Tracy, Gammon Luke, Liu Lu, Mellerio Jemima E, Dopping-Hepenstal Patricia J C, Pacy John, Elia George, Jeffery Rosemary, Leigh Irene M, Navsaria Harshad, McGrath John A
The Guy's, King's College and St Thomas' School of Medicine, Genetic Skin Disease Group, St John's Institute of Dermatology, Division of Genetics and Molecular Medicine, London, UK.
J Invest Dermatol. 2008 Sep;128(9):2179-89. doi: 10.1038/jid.2008.78. Epub 2008 Apr 3.
Recessive dystrophic epidermolysis bullosa (RDEB) is a severe inherited skin-blistering disorder caused by mutations in the COL7A1 gene that lead to reduced type-VII collagen and defective anchoring fibrils at the dermal-epidermal junction (DEJ). Presently there are no effective treatments for this disorder. Recent mouse studies have shown that intradermal injections of normal human fibroblasts can generate new human type-VII collagen and anchoring fibrils at the DEJ. To assess potential clinical benefits in humans, we gave single intradermal injections of allogeneic fibroblasts to five subjects with RDEB. We noted increased type-VII collagen at the DEJ at 2 weeks and at 3 months following injection and increased anchoring fibrils, although none of these had normal morphology. No adverse effects, clinical or immunopathologic, were noted. We believe the major effect of allogeneic fibroblasts is to increase the recipients' own COL7A1 mRNA levels with greater deposition of mutant type-VII collagen at the DEJ and formation of additional rudimentary anchoring fibrils. Nevertheless, this mutant protein may be partially functional and capable of increasing adhesion at the DEJ. This is the first study demonstrating that intradermal injections of allogeneic fibroblasts have therapeutic potential in human subjects with RDEB.
隐性营养不良性大疱性表皮松解症(RDEB)是一种严重的遗传性皮肤水疱病,由COL7A1基因突变引起,该突变导致VII型胶原蛋白减少以及真皮 - 表皮交界处(DEJ)的锚定原纤维缺陷。目前,这种疾病尚无有效的治疗方法。最近的小鼠研究表明,皮内注射正常人成纤维细胞可在DEJ处产生新的人类VII型胶原蛋白和锚定原纤维。为了评估对人类的潜在临床益处,我们对五名RDEB患者进行了皮内注射同种异体成纤维细胞。我们注意到注射后2周和3个月时,DEJ处的VII型胶原蛋白增加,锚定原纤维也增加,尽管这些都没有正常的形态。未观察到临床或免疫病理学方面的不良反应。我们认为同种异体成纤维细胞的主要作用是提高受体自身的COL7A1 mRNA水平,使突变的VII型胶原蛋白在DEJ处有更多沉积,并形成额外的原始锚定原纤维。然而,这种突变蛋白可能具有部分功能,能够增加DEJ处的黏附力。这是第一项证明皮内注射同种异体成纤维细胞对RDEB患者具有治疗潜力的研究。