免疫治疗反应性神经病理性疼痛和感觉过敏症患者的甘氨酸受体自身抗体：病例报告。
Immunotherapy-Responsive Neuropathic Pain and Allodynia in a Patient With Glycine Receptor Autoantibodies: A Case Report.
机构信息
From the Department of Neurology (B.S.), Oxford University Hospitals; Department of Clinical Neurology (C.B.), John Radcliffe Hospital Oxford; Oxford Autoimmune Neurology Group (C.B., S.R.I.), Nuffield Department of Clinical Neurosciences University of Oxford; Department of Clinical Neurosciences (T.Y.), Royal Infirmary Edinburgh; Wellcome Centre for Integrative Neuroimaging (I.T.), University of Oxford; Department of Clinical Neurosciences (P.F.), Royal Infirmary of Edinburgh; and Anne Rowling Regenerative Neurology Clinic (P.F.), University of Edinburgh.
出版信息
Neurol Neuroimmunol Neuroinflamm. 2023 Aug 28;10(6). doi: 10.1212/NXI.0000000000200160. Print 2023 Nov.
OBJECTIVES
Neuropathic pain is common and distressing. Improved mechanistic understanding and pharmacotherapies are urgently needed. Molecularly specific pain syndromes may provide insights with translational relevance. Glycine receptors are known to play a key role in inhibitory neurotransmission in the spinal dorsal horn and have therefore been considered as targets for analgesic development. While autoantibodies directed against glycine receptors may rarely arise spontaneously in humans, a detailed phenotype of neuropathic pain and allodynia in association with these autoantibodies has not been described.
METHODS
We describe the case of a previously well adult presenting with severe neuropathic pain and allodynia as part of an autoimmune brainstem and spinal syndrome with glycine receptor autoantibodies.
RESULTS
Our patient experienced a severe illness, including marked neuropathic pain and allodynia, hypoventilation, tetraparesis, and ophthalmoplegia. A diagnosis of progressive encephalomyelitis with rigidity and myoclonus was made. Neuropathic pain was characterized with validated instruments and responded promptly to cause-directed immunotherapy.
DISCUSSION
A detailed longitudinal phenotyping, using validated pain measurement instruments, of severe neuropathic pain and allodynia associated with likely pathogenic glycine receptor autoantibodies is reported. This case may have relevance for translational development of analgesics targeting glycinergic neurotransmission.
目的
神经病理性疼痛很常见且令人痛苦。迫切需要更好地了解其发病机制并研发出更多的治疗药物。具有分子特异性的疼痛综合征可能为具有转化相关性的研究提供新的思路。众所周知，甘氨酸受体在脊髓背角的抑制性神经传递中起着关键作用，因此被认为是开发镇痛药物的靶点。虽然针对甘氨酸受体的自身抗体在人类中很少自发产生，但与这些自身抗体相关的神经病理性疼痛和感觉过敏的详细表型尚未描述。
方法
我们描述了一例先前健康的成年患者，其表现为严重的神经病理性疼痛和感觉过敏，作为自身免疫性脑干和脊髓综合征的一部分，伴有甘氨酸受体自身抗体。
结果
我们的患者经历了严重的疾病，包括明显的神经病理性疼痛和感觉过敏、呼吸不足、四肢瘫痪和眼肌麻痹。诊断为进行性脑脊髓炎伴僵硬和肌阵挛。使用验证过的工具对神经病理性疼痛进行了详细的纵向表型分析，并对其进行了针对性免疫治疗，疼痛迅速缓解。
讨论
本文报告了一例严重的神经病理性疼痛和感觉过敏与可能的致病性甘氨酸受体自身抗体相关的详细纵向表型，使用了验证过的疼痛测量工具。该病例可能与靶向甘氨酸能神经传递的镇痛药物的转化发展有关。
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