Autoantibodies Directed Against Insulin Receptor During the Course of Castleman Disease: A New Case Reaffirming Autoimmune Hypoglycemia as a Relapse Warning Signal.

Q3 Medicine

European journal of case reports in internal medicine Pub Date : 2025-04-29 eCollection Date: 2025-01-01 DOI:10.12890/2025_005355

Xavier Jannot, Martine Auclair, Martin Martinot, Amin Maazouzi, David Boutboul, Corinne Vigouroux, Yannick Dieudonné

{"title":"Autoantibodies Directed Against Insulin Receptor During the Course of Castleman Disease: A New Case Reaffirming Autoimmune Hypoglycemia as a Relapse Warning Signal.","authors":"Xavier Jannot, Martine Auclair, Martin Martinot, Amin Maazouzi, David Boutboul, Corinne Vigouroux, Yannick Dieudonné","doi":"10.12890/2025_005355","DOIUrl":null,"url":null,"abstract":"This case study presents the case of a 54-year-old human immunodeficiency virus (HIV)-positive male who developed type B insulin resistance syndrome (TBIRS) in conjunction with a relapse of human herpesvirus 8 (HHV8)-positive multicentric Castleman disease (MCD). This case is only the sixth reported instance of TBIRS associated with HHV8-associated MCD. The diagnosis was confirmed by the presence of anti-insulin receptor autoantibodies, and the patient was treated effectively with rituximab, with no relapse in follow-up. The cases described are discussed, along with the differences between them and our own case. Additionally, the potential for an autoimmune complication of MCD, even when HIV is well controlled, is addressed, as well as the available therapeutic approaches.Learning points: Unexplained hypoglycemia can reveal autoimmunity against insulin receptors associated with lymphoproliferative disorders, including multicentric Castleman disease.Autoimmune hypoglycemia can occur independently of inflammatory signs or uncontrolled human immunodeficiency virus infection in patients with multicentric Castleman disease.","PeriodicalId":11908,"journal":{"name":"European journal of case reports in internal medicine","volume":"12 5","pages":"005355"},"PeriodicalIF":0.0000,"publicationDate":"2025-04-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12061218/pdf/","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"European journal of case reports in internal medicine","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.12890/2025_005355","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/1/1 0:00:00","PubModel":"eCollection","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}

引用次数: 0

Abstract

This case study presents the case of a 54-year-old human immunodeficiency virus (HIV)-positive male who developed type B insulin resistance syndrome (TBIRS) in conjunction with a relapse of human herpesvirus 8 (HHV8)-positive multicentric Castleman disease (MCD). This case is only the sixth reported instance of TBIRS associated with HHV8-associated MCD. The diagnosis was confirmed by the presence of anti-insulin receptor autoantibodies, and the patient was treated effectively with rituximab, with no relapse in follow-up. The cases described are discussed, along with the differences between them and our own case. Additionally, the potential for an autoimmune complication of MCD, even when HIV is well controlled, is addressed, as well as the available therapeutic approaches.

Learning points: Unexplained hypoglycemia can reveal autoimmunity against insulin receptors associated with lymphoproliferative disorders, including multicentric Castleman disease.Autoimmune hypoglycemia can occur independently of inflammatory signs or uncontrolled human immunodeficiency virus infection in patients with multicentric Castleman disease.

查看原文本刊更多论文

Castleman病过程中针对胰岛素受体的自身抗体：一个新病例重申自身免疫性低血糖是复发警告信号。

本病例研究报告了一名54岁的人类免疫缺陷病毒（HIV）阳性男性，他发展为B型胰岛素抵抗综合征（TBIRS），并伴有人类疱疹病毒8 （HHV8）阳性多中心Castleman病（MCD）的复发。该病例仅是报道的第六例与hhv8相关MCD相关的TBIRS病例。通过抗胰岛素受体自身抗体的存在证实了诊断，患者接受了有效的利妥昔单抗治疗，随访中无复发。讨论了所描述的案例，以及它们与我们自己的案例之间的区别。此外，即使在HIV得到良好控制的情况下，MCD的自身免疫性并发症的可能性也得到了解决，以及可用的治疗方法。学习要点：不明原因的低血糖可以揭示与淋巴增生性疾病（包括多中心Castleman病）相关的胰岛素受体自身免疫。自身免疫性低血糖可在多中心Castleman病患者中独立于炎症体征或不受控制的人类免疫缺陷病毒感染而发生。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

求助全文

约1分钟内获得全文求助全文

来源期刊

European journal of case reports in internal medicine

CiteScore

2.10

自引率

0.00%

发文量

166

审稿时长

8 weeks

期刊介绍： The European Journal of Case Reports in Internal Medicine is an official journal of the European Federation of Internal Medicine (EFIM), representing 35 national societies from 33 European countries. The Journal''s mission is to promote the best medical practice and innovation in the field of acute and general medicine. It also provides a forum for internal medicine doctors where they can share new approaches with the aim of improving diagnostic and clinical skills in this field. EJCRIM welcomes high-quality case reports describing unusual or complex cases that an internist may encounter in everyday practice. The cases should either demonstrate the appropriateness of a diagnostic/therapeutic approach, describe a new procedure or maneuver, or show unusual manifestations of a disease or unexpected reactions. The Journal only accepts and publishes those case reports whose learning points provide new insight and/or contribute to advancing medical knowledge both in terms of diagnostics and therapeutic approaches. Case reports of medical errors, therefore, are also welcome as long as they provide innovative measures on how to prevent them in the current practice (Instructive Errors). The Journal may also consider brief and reasoned reports on issues relevant to the practice of Internal Medicine, as well as Abstracts submitted to the scientific meetings of acknowledged medical societies.