Monozygotic twins with MELAS-like syndrome lacking ragged red fibers and lactacidaemia

被引:5
作者
Melberg, A
Akerlund, P
Raininko, R
Silander, HC
Wibom, R
Khaled, A
Nennesmo, I
Lundberg, PO
Olsson, Y
机构
[1] UNIV UPPSALA HOSP,DEPT NEURORADIOL,S-75185 UPPSALA,SWEDEN
[2] UNIV UPPSALA HOSP,DEPT NEUROSURG,S-75185 UPPSALA,SWEDEN
[3] UNIV UPPSALA HOSP,DEPT PATHOL,S-75185 UPPSALA,SWEDEN
[4] FALUN CENT HOSP,DEPT GERIATR & REHABIL,FALUN,SWEDEN
[5] HUDDINGE UNIV HOSP,DEPT CLIN CHEM,S-14186 HUDDINGE,SWEDEN
[6] HUDDINGE UNIV HOSP,DEPT PATHOL,S-14186 HUDDINGE,SWEDEN
来源
ACTA NEUROLOGICA SCANDINAVICA | 1996年 / 94卷 / 04期
关键词
MELAS; twins; focal status epilepticus; epilepsia partialis continua; generalised seizures; stroke-like episodes; mitochondrial angiopathy; complex I;
D O I
10.1111/j.1600-0404.1996.tb07058.x
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Typical cases of MELAS present a combination of clinical and neuroradiological features, lactacidaemia, and ragged red fibers (RRFs) in striated muscle. We have observed a MELAS-like syndrome in monozygotic twins. They developed seizures typically in conjunction with physical exertion, sleep deprivation or febrile episodes. Stroke-like episodes occurred usually during seizures. In twin 2 the course was fatal at age 20 years. Neuroradiological findings were typical of MELAS. Plasma lactate was normal in both. CSF lactate was normal in twin 1 and normal/elevated in twin 2. RRFs were not seen in muscle biopsies of the twins. Complex I activity was reduced in muscle in twin 1. Brain tissue removed at epilepsy surgery in twin 2 showed the presence of mitochondrial angiopathy. The commonest mitochondrial DNA mutation in MELAS, at base pair 3243, was absent. Lactacidaemia and mitochondrial myopathy with RRFs constitute part of the diagnostic criteria of MELAS. However, the absence of these features does not exclude mitochondrial disorder with the serious manifestations of MELAS (seizures and stroke-like episodes) as seen in these twins.
引用
收藏
页码:233 / 241
页数:9
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