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小儿运动性炎性神经病:抗磷脂抗体的作用

Pediatric Motor Inflammatory Neuropathy: The Role of Antiphospholipid Antibodies.

作者信息

Brogna Claudia, Luigetti Marco, Norcia Giulia, Scalise Roberta, Ferrantini Gloria, Berti Beatrice, Romeo Domenico M, Manna Raffaele, Mercuri Eugenio, Pane Marika

机构信息

Pediatric Neurology and Nemo Clinical Centre, Fondazione Policlinico Universitario A. Gemelli IRCSS, Università Cattolica del Sacro Cuore, Roma 00168, Italy.

Pediatric Neuropsichiatric Unit, ASL, Avellino 83100, Italy.

出版信息

Brain Sci. 2020 Mar 7;10(3):156. doi: 10.3390/brainsci10030156.

DOI:10.3390/brainsci10030156
PMID:32156084
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7139660/
Abstract

We report the clinical case of a nine-year-old girl who presented with progressive motor neuropathy, revealed via the detection of a higher delay in F-wave recording using digitalized nerve conduction/electromyography. Since the lupus anticoagulant (LAC) positivity, detected using diluted Russell viper venom time (dRVVT), switched to persistent serological anticardiolipin immunoglobulin G (IgG) positivity, a possible non-thrombotic antiphospholipid antibody (aPL)-related clinical manifestation was suspected, and intravenous immunoglobulin treatment (IVIG) was started. The IVIG treatment was well tolerated and the complete resolution of motor impairment was obtained after the third IVIG infusion. Our findings suggest that it could be useful to check for antiphospholipid antibodies in children with a rapid onset of progressive neurological signs in order to provide the beneficial use of IVIG in the treatment of pediatric aPL neurological conditions.

摘要

我们报告了一名9岁女孩的临床病例,该女孩表现为进行性运动神经病,通过数字化神经传导/肌电图检测发现F波记录延迟增加得以揭示。由于使用稀释的蝰蛇毒时间(dRVVT)检测到狼疮抗凝物(LAC)阳性转变为持续性血清抗心磷脂免疫球蛋白G(IgG)阳性,怀疑可能存在非血栓性抗磷脂抗体(aPL)相关的临床表现,并开始进行静脉注射免疫球蛋白治疗(IVIG)。IVIG治疗耐受性良好,在第三次IVIG输注后运动障碍完全缓解。我们的研究结果表明,对于快速出现进行性神经体征的儿童,检查抗磷脂抗体可能有用,以便在治疗儿童aPL神经系统疾病中有益地使用IVIG。

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本文引用的文献

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European evidence-based recommendations for diagnosis and treatment of paediatric antiphospholipid syndrome: the SHARE initiative.欧洲基于证据的儿科抗磷脂综合征诊断和治疗建议:SHARE 倡议。
Ann Rheum Dis. 2017 Oct;76(10):1637-1641. doi: 10.1136/annrheumdis-2016-211001. Epub 2017 May 4.
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Motor neuropathies and lower motor neuron syndromes.运动神经病和下运动神经元综合征
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Dev Med Child Neurol. 2011 Jun;53(6):522-8. doi: 10.1111/j.1469-8749.2011.03922.x.
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Six-Minute Walk Test demonstrates motor fatigue in spinal muscular atrophy.六分钟步行试验可显示出脊髓性肌萎缩症的运动疲劳。
Neurology. 2010 Mar 9;74(10):833-8. doi: 10.1212/WNL.0b013e3181d3e308.
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Antiphospholipid antibodies in pediatric systemic lupus erythematosus and the antiphospholipid syndrome.儿童系统性红斑狼疮和抗磷脂综合征中的抗磷脂抗体
Lupus. 2007;16(8):627-33. doi: 10.1177/0961203307079036.