Egger J, Lake B D, Wilson J
Arch Dis Child. 1981 Oct;56(10):741-52. doi: 10.1136/adc.56.10.741.
Thirteen children with abnormal mitochondria in muscle tissue, and a progressive neurological disorder that affected the cerebrum, cerebellum, extrapyramidal system, vestibular system, retina, upper motor neuron, lower motor neuron, and musculature, are reported. Other signs and symptoms were short stature, diabetes mellitus, cardiopathy, hypoplastic anaemia, glomerulopathy, and renal tubular dysfunction. These symptoms may occur singly or in various combinations and the manifestation may differ even within the same family. The most common clinical picture was that of "ophthalmoplegia plus'. Occurrence in relatives varied from isolated symptoms to the complete syndrome with "ragged red fibres' and is not inconsistent with an autosomal dominant mode of inheritance with variable expressivity. Theories for the pathophysiological basis of this syndrome are discussed and the literature reviewed.
报告了13名肌肉组织中线粒体异常且患有进行性神经疾病的儿童,该疾病影响大脑、小脑、锥体外系、前庭系统、视网膜、上运动神经元、下运动神经元和肌肉组织。其他体征和症状包括身材矮小、糖尿病、心脏病、发育不全性贫血、肾小球病和肾小管功能障碍。这些症状可能单独出现或多种组合出现,甚至在同一家族中表现也可能不同。最常见的临床症状是“眼肌麻痹加”。亲属中的发病情况从孤立症状到伴有“破碎红纤维”的完全综合征不等,这与具有可变表达性的常染色体显性遗传模式并不矛盾。本文讨论了该综合征病理生理基础的理论并回顾了相关文献。