Yang Yulong, Jiang Hailin, Yang Wenming, Wang Han, Wang Meixia, Li Xiang, Huang Peng, Fang Shuzhen, Hao Wenjie, Yang Yue, Zhao Furong, He Wei
Department of Neurology, The First Affiliated Hospital of Anhui University of Traditional, Chinese Medicine, Hefei, Anhui, China.
Key Laboratory of Xin'An Medicine, Ministry of Education Hefei, Anhui, China.
Front Immunol. 2025 Jan 7;15:1471115. doi: 10.3389/fimmu.2024.1471115. eCollection 2024.
Autoimmune polyendocrine syndromes (APS) is a rare group of disorders caused by impaired function of multiple endocrine glands due to disruption of immune tolerance. Of which, type 2 (APS-2) is the most common. Glutamic acid decarboxylase (GAD) is the rate-limiting enzyme for the synthesis of gamma-aminobutyric acid (GABA). Anti-GAD antibodies are associated with various neurological disorders, including stiff person syndrome (SPS). SPS is characterized by axial muscle stiffness, rigidity, and intermittent painful muscle spasms, with a prevalence of one to two in a million, making it an extremely rare neurological disorder. The comorbidity of APS-2 with SPS is even rarer. Most practicing neurologists encounter only one or two cases of APS-2 combined with anti-GAD65 antibody-associated SPS in their careers, resulting in underdiagnosis and undertreatment, leading to severe disability and suffering. This case report describes a young male who initially exhibited hair loss, vitiligo, and previously unreported eosinophilia. Before his diagnosis, he was admitted multiple times, with symptoms improving following the addition of intravenous immunoglobulin (IVIG) therapy to a poor treatment regimen. This paper aims to increase physicians' awareness of this condition, enhancing the likelihood of early diagnosis and treatment.
自身免疫性多内分泌综合征(APS)是一组罕见的疾病,由免疫耐受破坏导致多个内分泌腺功能受损引起。其中,2型(APS-2)最为常见。谷氨酸脱羧酶(GAD)是γ-氨基丁酸(GABA)合成的限速酶。抗GAD抗体与多种神经系统疾病相关,包括僵人综合征(SPS)。SPS的特征是轴向肌肉僵硬、强直和间歇性疼痛性肌肉痉挛,患病率为百万分之一至二,使其成为一种极其罕见的神经系统疾病。APS-2与SPS的合并症更为罕见。大多数执业神经科医生在其职业生涯中仅遇到一两个APS-2合并抗GAD65抗体相关SPS的病例,导致诊断不足和治疗不足,从而导致严重残疾和痛苦。本病例报告描述了一名年轻男性,他最初表现为脱发、白癜风和先前未报告的嗜酸性粒细胞增多。在确诊之前,他多次住院,在不良治疗方案中加用静脉注射免疫球蛋白(IVIG)治疗后症状有所改善。本文旨在提高医生对这种疾病的认识,增加早期诊断和治疗的可能性。