Department of Pediatrics and Adolescent Medicine, First Faculty of Medicine, Charles University in Prague and General University Hospital in Prague, Czech Republic.
Mol Genet Metab. 2013 Jan;108(1):102-5. doi: 10.1016/j.ymgme.2012.11.002. Epub 2012 Nov 13.
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.
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。