Faculty of Medicine of Sfax, Human Molecular Genetic Laboratory, University of Sfax, Tunisia.
Biochem Biophys Res Commun. 2012 Dec 7;429(1-2):31-8. doi: 10.1016/j.bbrc.2012.10.105. Epub 2012 Nov 3.
Pompe disease is a progressive metabolic myopathy caused by deficiency in lysosomal acid α-glucosidase and results in cellular lysosomal and cytoplasmic glycogen accumulation. A wide spectrum of clinical phenotypes exists from hypotonia and severe cardiac hypertrophy in the first few months of life to a milder form with the onset of symptoms in adulthood. The disease is typically due to severe mutations in GAA gene. In the present study, we described a newborn boy with clinical features of Pompe disease particularly with hypertrophic cardiomyopathy, hypotonia and hepatomegaly. This case was at first misdiagnosed as mitochondrial disorder. Accordingly, we performed a mitochondrial mutational analysis that revealed a novel mutation m.12908T>A in the ND5 gene. Secondary structure analysis of the ND5 protein further supported the deleterious role of the m.12908T>A mutation, as it was found to involve an extended imbalance in its hydrophobicity and affect its function.
庞贝病是一种由溶酶体酸性α-葡萄糖苷酶缺乏引起的进行性代谢性肌病,导致细胞溶酶体和细胞质糖原积累。从出生后数月内出现的肌张力低下和严重的心肌肥厚到成年后发病的较轻形式,存在广泛的临床表型。该病通常是由于 GAA 基因的严重突变引起的。在本研究中,我们描述了一名新生儿男孩,具有庞贝病的临床特征,特别是肥厚型心肌病、肌张力低下和肝肿大。该病例最初被误诊为线粒体疾病。因此,我们进行了线粒体突变分析,发现了 ND5 基因中的一个新突变 m.12908T>A。ND5 蛋白的二级结构分析进一步支持了 m.12908T>A 突变的有害作用,因为它涉及到疏水性的不平衡扩展,并影响其功能。