Sakai Norio, Otomo Takanobu
Child Healthcare and Genetic Science Laboratory, Division of Health Science, Osaka University Graduate School of Medicine, Osaka, Japan.
Department of Genetics, Osaka University Graduate School of Medicine, Osaka, Japan.
J Neurosci Res. 2016 Nov;94(11):1025-30. doi: 10.1002/jnr.23914.
Krabbe disease is an autosomal recessive, inherited demyelinating disease caused by deficiency of the lysosomal enzyme galactocerebrosidase. It is recognized as one of the predominant genetic diseases showing leukodystrophy from infancy to adulthood. The clinical phenotype and genotype for this disease show considerable variation worldwide, which makes accurate diagnosis difficult. Effective therapy is limited, although hematopoietic stem cell transplantation at an early stage has been established to some extent. We report here the long-term clinical effect on juvenile Krabbe disease for two brothers who underwent hematopoietic stem cell transplantation at an early stage of their disease. We review research into genotype-phenotype correlation for the possibility of early diagnosis at a presymptomatic stage. Medical care for this intractable disease will improve in the near future as a result of the increasing awareness of its molecular pathology and improvements in medical treatment. © 2016 Wiley Periodicals, Inc.
克拉伯病是一种常染色体隐性遗传性脱髓鞘疾病,由溶酶体酶半乳糖脑苷脂酶缺乏引起。它被认为是从婴儿期到成年期表现为脑白质营养不良的主要遗传性疾病之一。该疾病的临床表型和基因型在全球范围内存在相当大的差异,这使得准确诊断变得困难。尽管早期造血干细胞移植在一定程度上已得到确立,但有效治疗方法仍然有限。我们在此报告了两名患有幼年型克拉伯病的兄弟在疾病早期接受造血干细胞移植后的长期临床效果。我们回顾了关于基因型 - 表型相关性的研究,探讨在症状前阶段进行早期诊断的可能性。由于对其分子病理学的认识不断提高以及医疗治疗的改进,这种难治性疾病的医疗护理在不久的将来将会得到改善。© 2016 威利期刊公司