Department of Neurology, School of Mental Health and Neuroscience, Maastricht University Medical Center, Maastricht, The Netherlands.
Department of Clinical Genetics, Maastricht University Medical Center, Maastricht, The Netherlands.
PLoS One. 2016 Feb 11;11(2):e0148316. doi: 10.1371/journal.pone.0148316. eCollection 2016.
Screening for Fabry disease in patients with small fiber neuropathy has been suggested, especially since Fabry disease is potentially treatable. However, the diagnostic yield of testing for Fabry disease in isolated small fiber neuropathy patients has never been systematically investigated. Our aim is to determine the presence of Fabry disease in patients with small fiber neuropathy.
Patients referred to our institute, who met the criteria for isolated small fiber neuropathy were tested for Fabry disease by measurement of alpha-Galactosidase A activity in blood, lysosomal globotriaosylsphingosine in urine and analysis on possible GLA gene mutations.
725 patients diagnosed with small fiber neuropathy were screened for Fabry disease. No skin abnormalities were seen except for redness of the hands or feet in 30.9% of the patients. Alfa-Galactosidase A activity was tested in all 725 patients and showed diminished activity in eight patients. Lysosomal globotriaosylsphingosine was examined in 509 patients and was normal in all tested individuals. Screening of GLA for mutations was performed for 440 patients, including those with diminished α-Galactosidase A activity. Thirteen patients showed a GLA gene variant. One likely pathogenic variant was found in a female patient. The diagnosis Fabry disease could not be confirmed over time in this patient. Eventually none of the patients were diagnosed with Fabry disease.
In patients with isolated small fiber neuropathy, and no other signs compatible with Fabry disease, the diagnostic yield of testing for Fabry disease is extremely low. Testing for Fabry disease should be considered only in cases with additional characteristics, such as childhood onset, cardiovascular disease, renal failure, or typical skin lesions.
已建议对小纤维神经病患者进行法布里病筛查,尤其是因为法布里病是可治疗的。然而,尚未系统研究在孤立性小纤维神经病患者中检测法布里病的诊断收益。我们的目的是确定小纤维神经病患者中是否存在法布里病。
我们对符合孤立性小纤维神经病标准的我院就诊患者进行法布里病检测,方法是测量血液中的α-半乳糖苷酶 A 活性、尿液中的溶酶体神经酰胺三己糖苷和分析可能的 GLA 基因突变。
筛查了 725 例诊断为小纤维神经病的患者是否患有法布里病。除 30.9%的患者手脚发红外,未见皮肤异常。对所有 725 名患者进行了α-半乳糖苷酶 A 活性检测,发现 8 名患者活性降低。对 509 名患者进行了溶酶体神经酰胺三己糖苷检测,所有受检者均正常。对包括α-半乳糖苷酶 A 活性降低患者在内的 440 名患者进行了 GLA 基因突变筛查。13 名患者携带 GLA 基因变异。在一名女性患者中发现了一种可能的致病性变异。但在该患者中,随着时间的推移,无法确认法布里病的诊断。最终,没有患者被诊断为法布里病。
在无其他与法布里病相符的体征的孤立性小纤维神经病患者中,检测法布里病的诊断收益极低。只有在有其他特征,如儿童期发病、心血管疾病、肾衰竭或典型皮肤病变的情况下,才应考虑检测法布里病。