Hsu Jung-Lung, Liao Ming-Feng, Hsu Hui-Ching, Weng Yi-Ching, Lo Ai-Lun, Chang Kuo-Hsuan, Chang Hong-Shiu, Kuo Hung-Chou, Huang Chin-Chang, Ro Long-Sun
Department of Neurology, Chang Gung Memorial Hospital, Linkou Medical Center and Chang Gung University College of Medicine, Taipei, Taiwan.
Taipei Medical University, Graduate Institute of Humanities in Medicine, Taipei, Taiwan.
PLoS One. 2017 Sep 28;12(9):e0183948. doi: 10.1371/journal.pone.0183948. eCollection 2017.
To investigate the clinical characteristics of patients with uncommon distal symmetric painful small-fiber neuropathy (DSPSFN).
From September 2012 to September 2014, participants between 18-70 years of age that had DSPSFN defined by clinical signs/symptoms and ID pain > 2 or DN4 > 4 on questionnaires for more than 1 month were included. Participants who had previous historical or laboratory evidence of common etiologies of DSPSFN were excluded. Enzyme activity and genetic studies for Fabry diseaseand familial amyloid polyneuropathy were performed after participants fulfilled the inclusion and exclusion criteria. The cryoglobulin test, autoantibodies studies and electrophysiological studies were performed in these participants.
In total, 100 cases were enrolled in the current study. Three cases of subclinical diabetes mellitus and two cases of fibromyalgia were found. Fabry disease (1%) and familial amyloid polyneuropathy (3%) with Ala97Ser transthyretin (TTR) mutations were also detected. The cryoglobulin test was positive in 30% of participants, and these participants had higher DN4 scores than the negative group. In the autoantibodies studies, 59% of the participants had abnormal anti-Ro/SSA and/or anti-La/SSB antibodies.
Cryoglobulinemia is not a rare etiology of uncommon DSPSFN. The long-term prognosis is quite good in these participants. From our structuralized protocol, Fabry disease and familial amyloid polyneuropathy could be easily detected in these cases of uncommon DSPSFN.
探讨罕见的远端对称性疼痛性小纤维神经病变(DSPSFN)患者的临床特征。
纳入2012年9月至2014年9月期间年龄在18至70岁之间、经临床体征/症状确诊为DSPSFN且在问卷上ID疼痛>2或DN4>4超过1个月的参与者。排除既往有DSPSFN常见病因的病史或实验室证据的参与者。在参与者符合纳入和排除标准后,进行法布里病和家族性淀粉样多神经病的酶活性及基因研究。对这些参与者进行冷球蛋白检测、自身抗体研究和电生理研究。
本研究共纳入100例患者。发现3例亚临床糖尿病和2例纤维肌痛。还检测到1%的法布里病和3%的伴有Ala97Ser转甲状腺素蛋白(TTR)突变的家族性淀粉样多神经病。30%的参与者冷球蛋白检测呈阳性,且这些参与者的DN4评分高于阴性组。在自身抗体研究中,59%的参与者抗Ro/SSA和/或抗La/SSB抗体异常。
冷球蛋白血症是罕见的DSPSFN的病因之一。这些参与者的长期预后相当良好。根据我们的结构化方案,在这些罕见的DSPSFN病例中可以很容易地检测到法布里病和家族性淀粉样多神经病。