Inbal A, Avissar N, Shaklai M, Kuritzky A, Schejter A, Ben-David E, Shanske S, Garty B Z
Division of Hematology, Beilinson Medical Center, Petah Tiqva, Israel.
Am J Med Genet. 1995 Jan 30;55(3):372-8. doi: 10.1002/ajmg.1320550325.
We describe 2 sibs (brother and sister) with myopathy, sideroblastic anemia, lactic acidosis, mental retardation, microcephaly, high palate, high philtrum, distichiasis, and micrognathia. Very low levels of cytochromes a, b, and c were detected in the patients' muscle mitochondria. Deposition of iron within the mitochondria of bone marrow erythroblasts was observed on electron microscopy. Irregular and enlarged mitochondria with paracrystalline inclusions were also seen on electron microscopy of the patients' muscle specimen. Examination of DNA from the affected sibs showed no deletions in the mitochondrial DNA nor the mutations identified in the syndromes of mitochondrial myopathy, encephalopathy, lactic acidosis, and strokelike episodes (MELAS) or myoclonus, and epilepsy associated with rugged-red fibers (MERRF). Since the parents were first cousins and 2 of 6 sibs (male and female) were affected, we suggest that the syndrome expressed by our patients represents a previously unknown autosomal recessive disorder that includes mitochondrial myopathy, lactic acidosis, and sideroblastic anemia.
我们描述了2名患有肌病、铁粒幼细胞性贫血、乳酸性酸中毒、智力发育迟缓、小头畸形、高腭弓、高鼻梁、双行睫和小颌畸形的同胞(兄妹)。在患者的肌肉线粒体中检测到细胞色素a、b和c的水平极低。电子显微镜观察发现骨髓成红细胞的线粒体中有铁沉积。在患者的肌肉标本电子显微镜检查中还发现了线粒体不规则且增大,伴有类晶体包涵体。对患病同胞的DNA检测显示,线粒体DNA没有缺失,也没有在线粒体肌病、脑病、乳酸性酸中毒和卒中样发作综合征(MELAS)或肌阵挛以及与破碎红纤维相关的癫痫(MERRF)中发现的突变。由于父母是近亲结婚,6名同胞中有2名(1男1女)患病,我们认为我们的患者所表现出的综合征代表一种先前未知的常染色体隐性疾病,包括线粒体肌病、乳酸性酸中毒和铁粒幼细胞性贫血。