Fine J-D, Johnson L B, Weiner M, Stein A, Cash S, Deleoz J, Devries D T, Suchindran C
National Epidermolysis Bullosa Registry, Nashville, Tennessee, USA.
J Hand Surg Br. 2005 Feb;30(1):14-22. doi: 10.1016/j.jhsb.2004.07.006.
Mitten deformities of the hands and feet occur in nearly every patient with the most severe subtype (Hallopeau-Siemens) of recessive dystrophic epidermolysis bullosa, and in at least 40-50% of all other recessive dystrophic epidermolysis bullosa patients. Smaller numbers of patients with dominant dystrophic, junctional, and simplex types of epidermolysis bullosa are also at risk of this complication. Surgical intervention is commonly performed to correct these deformities, but recurrence and the need for repeated surgery are common. Higher numbers of epidermolysis bullosa patients also develop musculoskeletal contractures in other anatomic sites, further impairing overall function. Lifetable analyses not only better project the cumulative risk of mitten deformities and other contractures but also emphasize the need for early surveillance and intervention, since both of these musculoskeletal complications may occur within the first year of life.
几乎每一位患有隐性营养不良型大疱性表皮松解症最严重亚型(Hallopeau-Siemens型)的患者都会出现手足连指畸形,在所有其他隐性营养不良型大疱性表皮松解症患者中,至少40%-50%也会出现该畸形。较少数量的显性营养不良型、交界型和单纯型大疱性表皮松解症患者也有发生这种并发症的风险。通常会进行手术干预来矫正这些畸形,但复发以及需要反复手术的情况很常见。更多的大疱性表皮松解症患者还会在其他解剖部位出现肌肉骨骼挛缩,进一步损害整体功能。寿命表分析不仅能更好地预测连指畸形和其他挛缩的累积风险,还强调了早期监测和干预的必要性,因为这两种肌肉骨骼并发症都可能在生命的第一年出现。