Bose Smriti, Jacob Saiju
Neurology, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK.
Neurology, University Hospitals Birmingham NHS Foundation Trust, Birmingham, UK
Pract Neurol. 2025 Jan 16;25(1):6-17. doi: 10.1136/pn-2023-003974.
Stiff-person syndrome (SPS) is an autoimmune disease associated mainly with antibodies to glutamic acid decarboxylase (GAD) or to glycine, characterised by intermittent painful spasms, stiffness and rigidity of the proximal and truncal muscles. Neuro-ophthalmological and gastrointestinal symptoms also occur. The symptoms are caused by neuronal excitability due to impaired inhibitory (gamma amino butyric acid [GABA] and glycine) neurotransmission. SPS is part of a larger spectrum of GAD antibody-spectrum disorders, which overlaps with autoimmune epilepsy, cerebellar ataxia, myoclonus, progressive encephalomyelitis, rigidity and myoclonus (PERM) and limbic encephalitis. PERM is often caused by antibodies against the glycine receptor. Some SPS cases are paraneoplastic. Diagnostic delay is often associated with irreversible disability, and therefore, clinicians need a high degree of clinical suspicion to make an earlier diagnosis. This review updates the various clinical presentations that should raise suspicion of SPS and its related conditions and includes a diagnostic algorithm and various treatment strategies including immunotherapy and GABA-ergic drugs.
僵人综合征(SPS)是一种主要与谷氨酸脱羧酶(GAD)或甘氨酸抗体相关的自身免疫性疾病,其特征为近端和躯干肌肉间歇性疼痛性痉挛、僵硬和强直。还会出现神经眼科和胃肠道症状。这些症状是由于抑制性(γ-氨基丁酸[GABA]和甘氨酸)神经传递受损导致神经元兴奋性增加所致。SPS是更广泛的GAD抗体谱疾病的一部分,与自身免疫性癫痫、小脑共济失调、肌阵挛、进行性脑脊髓炎、强直和肌阵挛(PERM)以及边缘性脑炎重叠。PERM通常由抗甘氨酸受体抗体引起。一些SPS病例为副肿瘤性。诊断延迟常与不可逆的残疾相关,因此,临床医生需要高度的临床怀疑以早期诊断。本综述更新了应引起对SPS及其相关疾病怀疑的各种临床表现,并包括诊断算法以及包括免疫疗法和GABA能药物在内的各种治疗策略。