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病例报告:儿童期起病的接触蛋白1抗体相关神经病变合并肾病及文献综述

Case Report: Pediatric age onset CNTN1 antibody-associated neuropathy with nephropathy and literature review.

作者信息

Bayraktar Eltutan Ceyda, Rinaldi Simon, Vural Atay, Aksu Bagdagul, Maras Genc Hülya, Pembegul Yildiz Edibe

机构信息

Division of Pediatric Neurology, Department of Pediatrics, Istanbul Faculty of Medicine, Istanbul University, Istanbul, Türkiye.

Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, University of Oxford, Oxford, United Kingdom.

出版信息

Front Immunol. 2025 Jun 18;16:1549363. doi: 10.3389/fimmu.2025.1549363. eCollection 2025.

DOI:10.3389/fimmu.2025.1549363
PMID:40607401
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12213591/
Abstract

We present a 12-year-old boy with acute onset sensorimotor neuropathy and membranous glomerulonephritis associated with contactin-1 antibodies. This prompted us to explore the clinical characteristics of this condition and assess whether its presentation differs between pediatric and adult patients. A comprehensive search was conducted across multiple online databases, including PubMed and EMBASE, using MeSH terms such as "chronic inflammatory demyelinating polyradiculopathy", "acute inflammatory demyelinating polyradiculopathy ", "CIDP", "Guillain Barre syndrome", "proteinuria", "nephrotic syndrome", "nephropathy", "renal disease", "glomerulonephritis", "membranous nephropathy", "autoimmune nodopathies", and "membranous glomerulonephritis". We reviewed publications up to October 2024 and identified 39 patients with anti-contactin associated CIDP (chronic inflammatory demyelinating polyradiculopathy) with membranous glomerulonephritis (MGN), including our case. This rare coexistence typically occurs at advanced ages, with only two pediatric cases. Clinical features were similar regardless of age at onset. We compared the onset, symptoms, progression, renal histopathology, and treatment responses between pediatric and adult patients.

摘要

我们报告了一名12岁男孩,患有急性起病的感觉运动性神经病和与接触蛋白-1抗体相关的膜性肾小球肾炎。这促使我们探究这种疾病的临床特征,并评估其在儿科和成人患者中的表现是否存在差异。我们使用“慢性炎症性脱髓鞘性多发性神经根神经病”“急性炎症性脱髓鞘性多发性神经根神经病”“慢性炎症性脱髓鞘性多发性神经根神经病(CIDP)”“吉兰-巴雷综合征”“蛋白尿”“肾病综合征”“肾病”“肾脏疾病”“肾小球肾炎”“膜性肾病”“自身免疫性结节病”和“膜性肾小球肾炎”等医学主题词(MeSH),在包括PubMed和EMBASE在内的多个在线数据库中进行了全面检索。我们回顾了截至2024年10月的出版物,确定了39例患有抗接触蛋白相关慢性炎症性脱髓鞘性多发性神经根神经病(CIDP)并伴有膜性肾小球肾炎(MGN)的患者,包括我们的病例。这种罕见的共存情况通常发生在老年,仅有两例儿科病例。无论发病年龄如何,临床特征相似。我们比较了儿科和成人患者的发病情况、症状、病情进展、肾脏组织病理学和治疗反应。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dded/12213591/ca129b77e269/fimmu-16-1549363-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dded/12213591/b99462ba9bb9/fimmu-16-1549363-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dded/12213591/ca129b77e269/fimmu-16-1549363-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dded/12213591/b99462ba9bb9/fimmu-16-1549363-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/dded/12213591/ca129b77e269/fimmu-16-1549363-g002.jpg

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本文引用的文献

1
Characteristics of anti-contactin1 antibody positive autoimmune nodopathies combined with membranous nephropathy.抗接触蛋白 1 抗体阳性自身免疫性结节病合并膜性肾病的特征。
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2
Anti-contactin 1 Antibody-associated Membranous Nephropathy in Chronic Inflammatory Demyelinating Polyneuropathy with Several Autoantibodies.慢性炎症性脱髓鞘性多发性神经病伴多种自身抗体中的抗接触蛋白1抗体相关膜性肾病
Intern Med. 2024;63(5):699-705. doi: 10.2169/internalmedicine.2126-23.
3
A rare case of membranous nephropathy associated with chronic inflammatory demyelinating polyradiculoneuropathy.
一例罕见的膜性肾病合并慢性炎症性脱髓鞘性多发性神经根神经病。
Ren Fail. 2023 Dec;45(1):2209659. doi: 10.1080/0886022X.2023.2209659.
4
CIDP/autoimmune nodopathies with nephropathy: a case series study.CIDP/自身免疫性神经节病伴肾病:病例系列研究。
Ann Clin Transl Neurol. 2023 May;10(5):706-718. doi: 10.1002/acn3.51754. Epub 2023 Mar 17.
5
Contactin-1 links autoimmune neuropathy and membranous glomerulonephritis.联系蛋白-1 将自身免疫性神经病和膜性肾小球肾炎联系起来。
PLoS One. 2023 Mar 9;18(3):e0281156. doi: 10.1371/journal.pone.0281156. eCollection 2023.
6
Chronic inflammatory demyelinating polyradiculoneuropathy concomitant with nephropathy.伴有肾病的慢性炎症性脱髓鞘性多发性神经根神经病。
Neurol Sci. 2022 Oct;43(10):5885-5898. doi: 10.1007/s10072-022-06215-4. Epub 2022 Jun 23.
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