Autoantibodies directed against insulin receptor during the course of Castleman disease: a new case reaffirming autoimmune hypoglycemia as a relapse warning signal

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...

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Published inEuropean journal of case reports in internal medicine Vol. 12; no. 5; p. 005355
Main Authors Jannot, Xavier, Auclair, Martine, Martinot, Martin, Maazouzi, Amin, Boutboul, David, Vigouroux, Corinne, Dieudonné, Yannick
Format Journal Article
LanguageEnglish
Published Italy SMC Media Srl 2025
SMC MEDIA SRL
Subjects
autoimmune disease
hypoglycemia
multicentric castleman disease
type b insulin resistance syndrome
Multicentric Castleman disease
type B insulin resistance syndrome
autoimmune disease
hypoglycemia
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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.
AbstractList 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.
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.
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.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.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.LEARNING POINTSUnexplained 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.
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. 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.
Author Jannot, Xavier
Auclair, Martine
Boutboul, David
Martinot, Martin
Dieudonné, Yannick
Vigouroux, Corinne
Maazouzi, Amin
AuthorAffiliation 1 Department of Clinical Immunology and Internal Medicine, National Reference Center for Systemic Autoimmune Diseases (CNR RESO), Tertiary Center for Primary Immunodeficiency, Strasbourg University Hospital, Strasbourg, France
7 Université de Strasbourg, INSERM UMR_S1109, Fédération Hospitalo-Universitaire OMICARE, Fédération de Médecine Translationnelle de Strasbourg (FMTS), Strasbourg, France
6 Department of Hematology, Hôpital Cochin, AP-HP, Université Paris Cité; National Reference Center for Castleman disease, Hôpital Saint Louis, AP-HP, Université Paris Cité, Paris, France
3 National Reference Centre for Rare Diseases of Insulin Secretion and Insulin Sensitivity (PRISIS), Department of Endocrinology, Diabetology and Reproductive Endocrinology, Assistance Publique–Hôpitaux de Paris, Saint Antoine Hospital, Paris, France
4 Sorbonne Université, INSERM UMRS_938, Saint-Antoine Research Centre, IHU ICAN, Paris, France
5 Department of Infectious Diseases, Hôpitaux Civils de Colmar, Colmar, F
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Cites_doi 10.1210/clinem/dgad125
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type B insulin resistance syndrome
autoimmune disease
hypoglycemia
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SubjectTerms autoimmune disease
hypoglycemia
multicentric castleman disease
type b insulin resistance syndrome
Title Autoantibodies directed against insulin receptor during the course of Castleman disease: a new case reaffirming autoimmune hypoglycemia as a relapse warning signal
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