Simon Eva, Wendel Udo, Schadewaldt Peter
Department of General Pediatrics, University Children's Hospital Düsseldorf, Düsseldorf, Germany.
Turk J Pediatr. 2005 Jan-Mar;47(1):8-13.
Maple syrup urine disease (MSUD) is a rare autosomal recessive disorder that causes acute and chronic brain dysfunction because of a neurotoxic effect of the accumulating branched chain amino acids (BCAA) and their corresponding keto acids. Aim of the treatment is a rapid reversal of the neonatal decompensation and a stable long-term metabolic control obtained by a carefully adjusted BCAA-low diet. In optimally treated patients, an unimpaired neurological and intellectual outcome is possible. Ten patients of Turkish origin suffering from MSUD are presently treated in the Metabolic Unit of the University Hospital in Düsseldorf, Germany. All patients show mild intellectual deficits; neurological impairment is rare. This paper aims to define the feasible standard of therapy and the resulting intellectual and psychosocial outcome achievable in MSUD patients of Turkish origin under high standard conditions of medical care for inborn errors of metabolism.
枫糖尿症(MSUD)是一种罕见的常染色体隐性疾病,由于累积的支链氨基酸(BCAA)及其相应的酮酸具有神经毒性作用,可导致急性和慢性脑功能障碍。治疗的目的是迅速扭转新生儿失代偿状态,并通过精心调整的低BCAA饮食实现稳定的长期代谢控制。在接受最佳治疗的患者中,有可能获得未受损的神经和智力发育结果。目前,德国杜塞尔多夫大学医院代谢科正在治疗10名患有MSUD的土耳其裔患者。所有患者均表现出轻度智力缺陷;神经损伤较为罕见。本文旨在确定在针对先天性代谢缺陷的高标准医疗条件下,土耳其裔MSUD患者可行的治疗标准以及由此可实现的智力和心理社会发育结果。