PDF(Pubmed)
Abstract：
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.
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.
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.
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.
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.
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.
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.
摘要：
目的：神经性疼痛是常见且令人痛苦的。迫切需要改进的机械理解和药物疗法。分子特异性疼痛综合征可以提供具有翻译相关性的见解。已知甘氨酸受体在脊髓背角的抑制性神经传递中起关键作用，因此被认为是镇痛发展的目标。虽然针对甘氨酸受体的自身抗体很少在人类中自发产生，尚未描述与这些自身抗体相关的神经性疼痛和异常性疼痛的详细表型。
方法：我们描述了一个先前患有严重神经性疼痛和异常性疼痛的成年人，这是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：我们的病人经历了严重的疾病，包括明显的神经性疼痛和异常性疼痛，通气不足,四肢轻瘫,和眼肌麻痹。诊断为进行性脑脊髓炎伴僵硬和肌阵挛症。神经性疼痛用经过验证的仪器进行表征，并迅速对原因导向的免疫疗法作出反应。
结论：详细的纵向表型分析，使用经过验证的疼痛测量仪器，据报道，严重的神经性疼痛和异常性疼痛与可能的致病性甘氨酸受体自身抗体相关。这种情况可能与靶向甘氨酸能神经传递的镇痛药的翻译发展有关。
方法：我们描述了一个先前患有严重神经性疼痛和异常性疼痛的成年人，这是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：我们的病人经历了严重的疾病，包括明显的神经性疼痛和异常性疼痛，通气不足,四肢轻瘫,和眼肌麻痹。诊断为进行性脑脊髓炎伴僵硬和肌阵挛症。神经性疼痛用经过验证的仪器进行表征，并迅速对原因导向的免疫疗法作出反应。
结论：详细的纵向表型分析，使用经过验证的疼痛测量仪器，据报道，严重的神经性疼痛和异常性疼痛与可能的致病性甘氨酸受体自身抗体相关。这种情况可能与靶向甘氨酸能神经传递的镇痛药的翻译发展有关。
