Venugopal B, Wong K T, Goto Y-I, Bhattacharjee M B
National Heart Institute, Kuala Lumpur, Malaysia.
Ultrastruct Pathol. 2006 May-Jun;30(3):135-41. doi: 10.1080/01913120600689624.
The authors describe the case of a 50-year-old man with chronic progressive external ophthalmoplegia (CPEO), diabetes mellitus (DM), and coronary artery disease. The patient had no cardiac conduction abnormalities. During coronary artery bypass surgery, his heart and two skeletal muscles were biopsied. All three muscles showed ragged red fibers. The heart muscle showed significant glycogen accumulation. Analysis of mitochondrial DNA (mtDNA) showed a 5019-base-pair deletion, with no duplications. There were morphologically abnormal mitochondria in all 3 muscles, with clinically apparent difference in preservation of function. The combination of diabetes mellitus and mtDNA deletion is fortuitous, as they can be causally linked. The cardiac pathology allows speculation about the possible adaptive processes that may occur in the heart in DM. There are few reported cases with CPEO and excess glycogen in the heart. Most show deposition of fat and poorer clinical outcomes as compared to those with glycogen deposition. This observation may lend support to the hypothesis that in the myocardium, adaptive responses are mediated via changes in glucose handling, whereas alterations in fat metabolism likely represent maladaptation.
作者描述了一名50岁男性的病例,该患者患有慢性进行性眼外肌麻痹(CPEO)、糖尿病(DM)和冠状动脉疾病。患者无心脏传导异常。在冠状动脉搭桥手术期间,对其心脏和两块骨骼肌进行了活检。所有三块肌肉均显示出破碎红纤维。心肌显示出明显的糖原积累。线粒体DNA(mtDNA)分析显示存在一个5019个碱基对的缺失,无重复。所有三块肌肉中的线粒体形态均异常,功能保留情况在临床上有明显差异。糖尿病和mtDNA缺失的合并是偶然的,因为它们可能存在因果联系。心脏病理学情况让人推测糖尿病患者心脏中可能发生的适应性过程。很少有CPEO且心脏中糖原过多的病例报道。与糖原沉积的病例相比,大多数病例表现为脂肪沉积且临床结局较差。这一观察结果可能支持以下假设:在心肌中,适应性反应是通过葡萄糖处理的变化介导的,而脂肪代谢的改变可能代表适应不良。