Mercimek-Mahmutoglu Saadet, Gruber Stephan, Rolfs Arndt, Stadlbauer Andreas, Woeber Christian, Kurnik Peter, Voigtlaender Till, Moser Ewald, Stoeckler-Ipsiroglu Sylvia
Department of Pediatrics, Medical University Vienna, Austria.
Mol Genet Metab. 2007 Aug;91(4):390-5. doi: 10.1016/j.ymgme.2007.03.009. Epub 2007 Jun 8.
Gaucher disease type 1 (GD1) is an autosomal recessive lysosomal storage disorder, characterised by accumulation of glycosphingolipids in visceral organs. Although considered non-neuronopathic neurological involvement has been reported in single cases. The aim of our study was to investigate central and peripheral nervous system involvement in patients with GD1. We investigated nine unrelated patients with GD1 by three-dimensional cerebral 1H-magnetic resonance spectroscopic imaging and clinical and neurophysiological tests. We found an increased choline level on MRS in four patients. One of these patients had mixed axonal neuropathy and subclinical involvement of the central somatosensory tract as well as monoclonal gammopathy. One patient with normal cerebral choline levels had evidence of bilateral carpal tunnel syndrome upon neurophysiological exam. The N370S mutation was found in 11 out of 18 alleles. Three patients were compound heterozygous for the L444P mutation. There was no correlation between increased cerebral choline levels and type of mutations. MRS findings suggest that in patients with classical non-neuronopathic GD1, the brain is involved at a subclinical level in some patients.
1型戈谢病(GD1)是一种常染色体隐性溶酶体贮积症,其特征为糖鞘脂在内脏器官中蓄积。尽管一般认为该病无神经病变,但已有个别病例报告出现神经受累情况。我们研究的目的是调查GD1患者的中枢和外周神经系统受累情况。我们通过三维脑1H磁共振波谱成像以及临床和神经生理学测试,对9例无亲缘关系的GD1患者进行了调查。我们发现4例患者磁共振波谱中的胆碱水平升高。其中1例患者存在混合性轴索性神经病、中枢体感束亚临床受累以及单克隆丙种球蛋白病。1例脑胆碱水平正常的患者经神经生理学检查有双侧腕管综合征的证据。在18个等位基因中,有11个发现了N370S突变。3例患者为L444P突变的复合杂合子。脑胆碱水平升高与突变类型之间无相关性。磁共振波谱结果表明,在典型的非神经病变型GD1患者中,部分患者的脑在亚临床水平受累。