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黏多糖贮积症Ⅰ型:骨髓移植后牙科检查结果5年随访的病例报告

Hurler syndrome: a case report of a 5-year follow-up of dental findings after bone marrow transplantation.

作者信息

Wadenya Rose O, Stout Angela M, Gupta Avin, Monge Janet

机构信息

Department of Preventive and Restorative Sciences, Division of Pediatric and Community Oral Health, University of Pennsylvania School of Dental Medicine, Philadelphia, Pennsylvania, USA.

出版信息

Spec Care Dentist. 2010 Jan-Feb;30(1):14-7. doi: 10.1111/j.1754-4505.2009.00115.x.

Abstract

Hurler syndrome is a rare autosomal recessive disorder of mucopolysaccharide metabolism. It results from a deficiency in lysosomal enzymes responsible for the breakdown of glycosaminoglycans. Affected individuals may show progressive physical and mental deterioration as glycosaminoglycans are deposited in the organs of the body. Bone marrow transplantation (BMT) is effective in improving some of the clinical manifestations of Hurler syndrome. Death is caused by cardiorespiratory failure and usually occurs before the second decade of life. In this case report, the course of dental development was followed over 5 years, from the primary dentition into the permanent dentition, of a child who was successfully treated with a bone marrow transplant in infancy. The timing of bone marrow therapy has significant and variable effect on the stages of tooth development with implications for the long-term maintenance of the dentition.

摘要

黏多糖贮积症Ⅰ型是一种罕见的常染色体隐性黏多糖代谢紊乱疾病。它是由负责分解糖胺聚糖的溶酶体酶缺乏引起的。由于糖胺聚糖沉积在身体器官中,受影响的个体可能会出现渐进性的身心衰退。骨髓移植(BMT)对于改善黏多糖贮积症Ⅰ型的一些临床表现有效。死亡由心肺衰竭引起,通常发生在生命的第二个十年之前。在本病例报告中,跟踪了一名婴儿期成功接受骨髓移植治疗的儿童从乳牙列到恒牙列5年的牙齿发育过程。骨髓治疗的时机对牙齿发育阶段有显著且可变的影响,这对牙列的长期维持具有重要意义。

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