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一个携带线粒体 MTATP6 基因 m.8851T>C 突变的家族中不同的实验室和肌肉活检结果。

Different laboratory and muscle biopsy findings in a family with an m.8851T>C mutation in the mitochondrial MTATP6 gene.

机构信息

Department of Pediatrics and Adolescent Medicine, First Faculty of Medicine, Charles University in Prague and General University Hospital in Prague, Czech Republic.

出版信息

Mol Genet Metab. 2013 Jan;108(1):102-5. doi: 10.1016/j.ymgme.2012.11.002. Epub 2012 Nov 13.

Abstract

We report the second known family with a very rare, maternally inherited missense m.8851T>C mutation in the mitochondrial MTATP6 gene. A failure to thrive, microcephaly, psychomotor retardation and hypotonia were present in a 3-year-old girl with a high mtDNA mutation load (87-97%). Ataxia and Leigh syndrome were subsequently documented in a neurological examination and brain MRI. A muscle biopsy demonstrated decreased ATP synthase and an accumulation of succinate dehydrogenase products, indicating mitochondrial myopathy. Her 36-year-old mother (68% blood heteroplasmy) developed peripheral neuropathy and muscle weakness at the age of 22 years. Our findings extend the clinical and laboratory phenotype associated with the m.8851T>C mutation.

摘要

我们报告了第二个已知的家族,该家族携带有非常罕见的母系遗传的线粒体 MTATP6 基因 m.8851T>C 错义突变。一名 3 岁女孩表现为生长不良、小头畸形、精神运动发育迟缓伴张力减退,其线粒体 DNA 突变负荷很高(87-97%)。随后的神经学检查和脑 MRI 显示共济失调和 Leigh 综合征。肌肉活检显示三磷酸腺苷合酶减少和琥珀酸脱氢酶产物堆积,提示线粒体肌病。她 36 岁的母亲(68%血液异质性)在 22 岁时出现周围神经病和肌肉无力。我们的发现扩展了与 m.8851T>C 突变相关的临床和实验室表型。

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