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部分而非所有类型黏多糖贮积症患者的毛发形态异常。

Abnormalities in the hair morphology of patients with some but not all types of mucopolysaccharidoses.

作者信息

Malinowska Marcelina, Jakóbkiewicz-Banecka Joanna, Kloska Anna, Tylki-Szymańska Anna, Czartoryska Barbara, Piotrowska Ewa, Wegrzyn Alicja, Wegrzyn Grzegorz

机构信息

Department of Molecular Biology, University of Gdańsk, Kładki 24, 80-822, Gdańsk, Poland.

出版信息

Eur J Pediatr. 2008 Feb;167(2):203-9. doi: 10.1007/s00431-007-0462-7. Epub 2007 Mar 15.

Abstract

Mucopolysaccharidoses (MPS) are a group of inherited, progressive, metabolic diseases, caused by the deficiency of one of the enzymes involved in the degradation of glycosaminoglycans (GAGs). The disease is usually fatal, with the life span of most untreated MPS patients being between one and two decades. In this report, on the basis of scanning electron microscopy (SEM) studies, we demonstrate that, besides the many other symptoms of MPS, there are characteristic abnormalities in the hair morphology of patients suffering from some types of this disease (MPS I, MPS II, MPS IIIA, MPS IIIB), but not from other types (MPS IVA, MPS IVB, MPS VI), where the changes are minor, if any. Different GAGs accumulate in the tissues of patients suffering from the various MPS types, and analysis of the disease types in which severe hair abnormalities occur or not could suggest that the accumulation of heparan sulfate, rather than dermatan sulfate or keratan sufate, may be responsible for the major changes in hair morphology. Considerable abnormalities in hair morphology occur in patients suffering from MPS I, MPS II, MPS IIIA, and MPS IIIB, but not in patients suffering from MPS IVA, MPS IVB, and MPS VI; this feature might potentially be used as an additional test for the assessment of the efficacy of treatments for MPS patients (types I, II, IIIA, and IIIB).

摘要

黏多糖贮积症(MPS)是一组遗传性、进行性代谢疾病,由参与糖胺聚糖(GAGs)降解的一种酶缺乏所致。该疾病通常是致命的,大多数未经治疗的MPS患者寿命在10至20年之间。在本报告中,基于扫描电子显微镜(SEM)研究,我们证明,除了MPS的许多其他症状外,患有某些类型该疾病(MPS I、MPS II、MPS IIIA、MPS IIIB)的患者毛发形态存在特征性异常,而其他类型(MPS IVA、MPS IVB、MPS VI)患者的毛发形态变化轻微或无变化。不同类型的GAGs在患有各种MPS类型的患者组织中蓄积,对出现或未出现严重毛发异常的疾病类型进行分析可能表明,硫酸乙酰肝素而非硫酸皮肤素或硫酸角质素的蓄积可能是毛发形态发生重大变化的原因。患有MPS I、MPS II、MPS IIIA和MPS IIIB的患者毛发形态出现明显异常,而患有MPS IVA、MPS IVB和MPS VI的患者则未出现;这一特征可能潜在地用作评估MPS患者(I、II、IIIA和IIIB型)治疗效果的额外检测方法。

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