Autoimmune hypoglycemia expands the biological spectrum of HHV8+ multicentric Castleman disease

Blood Adv. 2021 Apr 13;5(7):1848-1852. doi: 10.1182/bloodadvances.2020002801.

Pierre Arnautou¹, Martine Auclair², Soraya Fellahi³, Clara Bouché⁴, Claire Fieschi^{1

5}, Elias Barrak⁶, Viviane Queyrel-Moranne⁷, Lucy Chaillous⁶, Nicolas Blin⁶, Marion Malphettes^{1

5}, Jehane Fadlallah^{1

5}, Remi Bertinchamp^{1

5}, Laurence Gérard^{1

5}, Djaouida Bengoufa⁸, Lionel Galicier^{1

5}, Eric Oksenhendler^{1

5}, Corinne Vigouroux^{2

9

10}, David Boutboul^{1

5

11}

¹ Clinical Immunology Department, Hôpital Saint Louis, Université de Paris, Paris, France.
² Sorbonne Université, INSERM UMRS_938, Saint-Antoine Research Centre, IHU ICAN, Paris, France.
³ Biochemistry Laboratory, Hôpital Henri Mondor, Créteil, France.
⁴ Endocrinology Department, Hôpital Lariboisière, and.
⁵ National Reference Center for Castleman disease, Hôpital Saint Louis, Université de Paris, Paris, France.
⁶ Endocrinology Department, CHU Nantes, Nantes, France.
⁷ Rheumatology Department, CHU Nice, Nice, France.
⁸ Immunology and Histocompatibility Laboratory, Hôpital Saint Louis, Université de Paris, Paris, France.
⁹ Department of Endocrinology and.
¹⁰ Department of Molecular Biology and Genetics, Assistance Publique-Hôpitaux de Paris, Saint Antoine Hospital, National Reference Centre for Rare Diseases of Insulin Secretion and Insulin Sensitivity (PRISIS), Paris, France; and.
¹¹ U976 HIPI, INSIGHT team, Hôpital Saint Louis, Université de Paris, Paris, France.

Abstract

Autoimmune hypoglycemia belongs to the clinical spectrum of HHV8⁺ MCD and rituximab is an effective treatment of this condition.
This rare complication is related to autoantibodies directed toward the insulin receptor and activating the insulin signaling pathway.