Kurogouchi F, Oguchi T, Mawatari E, Yamaura S, Hora K, Takei M, Sekijima Y, Ikeda S i, Kiyosawa K
Second Department of Internal Medicine, Shinshu University School of Medicine, Matsumoto, Japan.
Am J Nephrol. 1998;18(6):551-6. doi: 10.1159/000013406.
A 27-year-old female with short stature and mild hearing loss was diagnosed as having focal-segmental glomerulosclerosis by renal biopsy at our hospital. One year later she developed progressive renal dysfunction and cardiac failure and was admitted again to our hospital for evaluation. Though her only neurological disorder was mild hearing loss, her short stature and elevated lactate and pyruvate values in cerebrospinal fluid suggested mitochondrial cytopathy. A muscle biopsy specimen of the left biceps brachii, using modified Gomori trichrome stain, showed a typical image of ragged-red fibers, and an increased number of giant mitochondria with paracrystalline inclusions were visible by electron microscopy. Mitochondrial DNA from the skeletal muscle showed an A-to-G transition at 3243 of transfer RNALeu(UUR), the common point mutation for mitochondrial myopathy, encephalopathy, lactic acidosis, and strokelike episodes. These data confirmed the diagnosis of atypical mitochondrial cytopathy with renal and heart involvement. Mitochondrial cytopathies are often associated with hypertrophic cardiomyopathy but rarely with renal disease. Among the few reported cases with associated renal disease, most included renal tubular disorders; few cases with focal glomerular sclerosis are known. The present case of atypical mitochondrial cytopathy was characterized by a unique clinical course and rare complications with focal-segmental glomerulosclerosis.
一名27岁身材矮小且伴有轻度听力损失的女性患者,在我院经肾活检被诊断为局灶节段性肾小球硬化症。一年后,她出现进行性肾功能不全和心力衰竭,并再次入院接受评估。尽管她唯一的神经功能障碍是轻度听力损失,但她的身材矮小以及脑脊液中乳酸和丙酮酸值升高提示线粒体细胞病。对左侧肱二头肌进行肌肉活检,采用改良的Gomori三色染色法,显示出线粒体肌病典型的破碎红纤维图像,通过电子显微镜可见大量含有副结晶包涵体的巨大线粒体。骨骼肌的线粒体DNA显示,在转运RNA亮氨酸(UUR)的3243位点发生了A到G的转变,这是线粒体肌病、脑病、乳酸酸中毒和卒中样发作的常见点突变。这些数据证实了该患者为伴有肾脏和心脏受累的非典型线粒体细胞病。线粒体细胞病常与肥厚型心肌病相关,但很少与肾脏疾病相关。在少数报道的伴有肾脏疾病的病例中,大多数包括肾小管疾病;已知伴有局灶性肾小球硬化的病例很少。本例非典型线粒体细胞病具有独特的临床病程和伴有局灶节段性肾小球硬化的罕见并发症。