Bastiaensen L A, Notermans S L, Ramaekers C H, van Dijke B J, Joosten E M, Jaspar H H, Stadhouders A M, Beljaars C T
Ophthalmologica. 1982;184(1):40-50. doi: 10.1159/000309183.
A 20-year-old man with the characteristic findings of infantile onset Kearns syndrome is described. Morphological and biochemical investigations proved a mitochondrial disease which we believe to be the cause of the symptoms in various organs. We assume an autosomal-dominant inheritance, the marker sign of which is blepharoptosis in several family members. Characteristic clinical, morphological and biochemical findings, combined with an autosomal-dominant inheritance with very variable expression, mark the Kearns syndrome as an individual disease, not as a symptom complex (syndrome). Kearns disease can be divided into three forms--an infantile form ("Kearns-Sayre syndrome') with early onset, rapid progression, multisystemic involvement and a severe course; and a juvenile and an adult form with onset in the second, respectively third (or later) decades with a generally slower and more benign course and less widespread expression in various organ systems. Furthermore, the occurrence of a curious orthoptic abnormality is described, indicating one of the possible ways to avoid diplopia in chronic progressive external ophthalmoplegia: the coexistence of normal and gliding abnormal retinal correspondence.
本文描述了一名患有婴儿期起病的卡恩斯综合征典型表现的20岁男性。形态学和生化检查证实为线粒体疾病,我们认为这是各器官症状的病因。我们推测其遗传方式为常染色体显性遗传,其标记性体征为多名家庭成员出现上睑下垂。典型的临床、形态学和生化表现,加上具有高度可变表达的常染色体显性遗传,使卡恩斯综合征成为一种独特的疾病,而非一组症状(综合征)。卡恩斯病可分为三种类型:婴儿型(“卡恩斯-塞尔综合征”)起病早、进展快、累及多系统且病情严重;青少年型和成人型分别在第二个十年及第三个十年(或更晚)起病,病程通常较缓慢且病情较轻,在各器官系统中的表现也较局限。此外,还描述了一种奇特的视轴矫正异常情况,这表明在慢性进行性外眼肌麻痹中避免复视的一种可能方式:正常视网膜对应与滑动性异常视网膜对应的共存。