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肾上腺脑白质营养不良:9名台湾儿童的临床分析

Adrenoleukodystrophy: clinical analysis of 9 Taiwanese children.

作者信息

Liang Jao-Shwann, Lee Wang-Tso, Hwu Wuh-Liang, Peng Steven Shinn-Forng, Chu Li-Wen, Wang Pen-Jung, Shen Yu-Zen

机构信息

Department of Pediatrics, Far Eastern Memorial Hospital, Panchiao City, Taipei 22050, Taiwan.

出版信息

Acta Paediatr Taiwan. 2004 Sep-Oct;45(5):272-7.

Abstract

X-linked adrenoleukodystrophy (X-ALD) is a peroxisomal disorder and identified in many races without apparent predilection for any race. This study was designed to investigate the clinical and therapeutic aspects of X-ALD in Taiwanese children with this disorder. We retrospectively reviewed all children admitted to NTUH from Nov. 1993 to Aug. 2002 with the diagnosis of ALD, defined by increased very long chain fatty acid (VLCFA). The mean age at diagnosis of the patients was 7.4 years (range, 2.8 to 13 years). Seven out of 9 patients had abnormal brain magnetic resonance image (MRI) studies. Three patients received bone marrow transplantation. Of these, two died of severe graft-versus-host disease and the other remained stable. Of the remaining 6 patients, two patients were in vegetative status and the other two patients were neurologically normal. X-ALD in Taiwanese children had similar clinical manifestations as reviewed in western countries. Symmetrical demyelination in parieto-occipital region and the accumulation of contrast material at the edge of the lesion are the typical MRI findings. Proton MR spectroscopy (MRS) can be used to evaluate either the asymptomatic patient or patient with normal brain image. Performance of T-cell depletion bone marrow transplantation or cord blood transplantation is suggested for X-ALD with early cerebral involvement.

摘要

X连锁肾上腺脑白质营养不良(X-ALD)是一种过氧化物酶体疾病,在许多种族中均有发现,无明显的种族倾向。本研究旨在调查台湾患有该疾病儿童的X-ALD临床及治疗情况。我们回顾性分析了1993年11月至2002年8月间入住台湾大学医院且诊断为ALD的所有儿童,ALD的诊断依据为极长链脂肪酸(VLCFA)升高。患者诊断时的平均年龄为7.4岁(范围为2.8至13岁)。9例患者中有7例脑部磁共振成像(MRI)检查异常。3例患者接受了骨髓移植。其中,2例死于严重的移植物抗宿主病,另1例病情稳定。其余6例患者中,2例处于植物人状态,另外2例神经功能正常。台湾儿童的X-ALD临床表现与西方国家报道的相似。顶枕区对称性脱髓鞘以及病变边缘对比剂积聚是典型的MRI表现。质子磁共振波谱(MRS)可用于评估无症状患者或脑部影像正常的患者。对于早期脑部受累的X-ALD患者,建议进行T细胞去除的骨髓移植或脐血移植。

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