Koo B, Becker L E, Chuang S, Merante F, Robinson B H, MacGregor D, Tein I, Ho V B, McGreal D A, Wherrett J R
Division of Neurology, Hospital for Sick Children, Toronto, Canada.
Ann Neurol. 1993 Jul;34(1):25-32. doi: 10.1002/ana.410340107.
We reviewed 10 patients (5 males, 5 females) with mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes. The age of symptom onset ranged from 3 months to 12 years. All had lactic acidosis, multiple stroke-like events with secondary neurological deficits, radiological changes of progressive brain infarction, and muscle biopsy showing ragged-red fibers. In patients with earlier onset of symptoms (< 2 yr), involvement tended to be more diffuse, with failure to thrive and early onset of delayed development. Patients whose symptoms appeared later tended to have focal neurological deficits with migraine-like headache, and a rate of cognitive regression reflecting the rapidity of disease progression. Radiological changes included multiple areas of infarction with initial predilection for parietal occipital areas, progressing to generalized atrophy. Pathological findings in muscle biopsies included type 1 fiber predominance, ragged-red fibers, increased intermyofibrillar lipid deposition, and abnormal mitochondria. Four patients showed mitochondrial DNA tRNA mutation at position 3,243. No difference was noted in clinical, radiological, or pathological findings in patients with and without this mutation, suggesting that multiple sites of point mutation may give rise to mitochondrial encephalomyopathy, lactic acidosis, and stroke-like episodes.
我们回顾了10例患有线粒体脑肌病、乳酸酸中毒和卒中样发作的患者(5例男性,5例女性)。症状出现的年龄范围为3个月至12岁。所有患者均有乳酸酸中毒、多次卒中样发作并伴有继发性神经功能缺损、进行性脑梗死的影像学改变以及肌肉活检显示破碎红纤维。症状出现较早(<2岁)的患者,受累往往更广泛,有生长发育迟缓及早期出现发育延迟。症状出现较晚的患者往往有局灶性神经功能缺损并伴有偏头痛样头痛,以及反映疾病进展速度的认知衰退率。影像学改变包括多个梗死区域,最初以顶枕叶区域为主,进而发展为广泛性萎缩。肌肉活检的病理结果包括1型纤维占优势、破碎红纤维、肌原纤维间脂质沉积增加以及线粒体异常。4例患者在3243位点出现线粒体DNA tRNA突变。有此突变和无此突变的患者在临床、影像学或病理结果上均未发现差异,这表明多个点突变位点可能导致线粒体脑肌病、乳酸酸中毒和卒中样发作。