Gonzalez C, Roth R
Otolaryngology Head and Neck Surgery Service, Walter Reed Army Medical Center, Washington, D.C. 20307.
Int J Pediatr Otorhinolaryngol. 1989 Jul;17(3):305-11. doi: 10.1016/0165-5876(89)90056-6.
Epidermolysis bullosa (EB) is a rare hereditary skin disease of infancy that can involve the mucous membranes of the oral cavity. Laryngotracheal involvement is rare. The disease is characterized by bullae formation in response to minor trauma. There are at least 18 described types of EB, however, there are 3 basic categories. These are simplex (with disruption above the basement membrane), dystrophic (in which disruption is below the basement membrane), and junctional (in which the split is within the lamina lucida). The prognosis of the different types ranges from early death usually secondary to overwhelming sepsis, to long term survivals with lack of growth retardation or significant dystrophic scarring. Presently, survival appears to be the only reliable criteria for distinguishing the benign and lethal forms of EB. Airway obstruction secondary to laryngotracheal involvement should be considered in any child with epidermolysis bullosa presented with symptoms of respiratory distress. Because there are few predictive prognostic indicators in the neonatal period, tracheotomy should be considered early in an effort to prevent further laryngeal injury from intubation, in those patients that will survive.
大疱性表皮松解症(EB)是一种罕见的婴儿遗传性皮肤病,可累及口腔黏膜。喉气管受累罕见。该病的特征是对轻微创伤产生水疱。EB至少有18种已描述的类型,然而,可分为3个基本类别。分别是单纯型(基底膜上方破裂)、营养不良型(破裂发生在基底膜下方)和交界型(分裂发生在透明层内)。不同类型的预后差异很大,从通常因严重败血症导致的早期死亡,到无生长发育迟缓或明显营养不良性瘢痕形成的长期存活。目前,生存似乎是区分EB良性和致死性形式的唯一可靠标准。任何患有大疱性表皮松解症且出现呼吸窘迫症状的儿童,都应考虑喉气管受累导致的气道梗阻。由于新生儿期几乎没有预测预后的指标,对于那些能够存活的患者,应尽早考虑气管切开术,以防止插管对喉部造成进一步损伤。