Diabetes and immune thrombocytopenic purpura: a new association with good response to anti-CD20 therapy.

Détails

ID Serval
serval:BIB_F096E98DD4AF
Type
Article: article d'un périodique ou d'un magazine.
Sous-type
Etude de cas (case report): rapporte une observation et la commente brièvement.
Collection
Publications
Titre
Diabetes and immune thrombocytopenic purpura: a new association with good response to anti-CD20 therapy.
Périodique
Pediatric Diabetes
Auteur(s)
von Laer Tschudin L., Schwitzgebel V.M., von Scheven-Gête A., Blouin J.L., Hofer M., Hauschild M., Ansari M., Stoppa-Vaucher S., Phan-Hug F.
ISSN
1399-5448 (Electronic)
ISSN-L
1399-543X
Statut éditorial
Publié
Date de publication
2015
Peer-reviewed
Oui
Volume
16
Numéro
2
Pages
138-145
Langue
anglais
Notes
Publication types: Journal Article Publication Status: ppublish, pdf: Case Report
Résumé
Type 1 diabetes (T1D) is rarely a component of primary immune dysregulation disorders. We report two cases in which T1D was associated with thrombocytopenia. The first patient, a 13-year-old boy, presented with immune thrombocytopenia (ITP), thyroiditis, and, 3 wk later, T1D. Because of severe thrombocytopenia resistant to immunoglobulins, high-dose steroids, and cyclosporine treatment, anti-cluster of differentiation (CD20) therapy was introduced, with consequent normalization of thrombocytes and weaning off of steroids. Three and 5 months after anti-CD20 therapy, levothyroxin and insulin therapy, respectively, were stopped. Ten months after stopping insulin treatment, normal C-peptide and hemoglobin A1c (HbA1c) levels and markedly reduced anti-glutamic acid decarboxylase (GAD) antibodies were measured. A second anti-CD20 trial for relapse of ITP was initiated 2 yr after the first trial. Anti-GAD antibody levels decreased again, but HbA1c stayed elevated and glucose monitoring showed elevated postprandial glycemia, demanding insulin therapy. To our knowledge, this is the first case in which insulin treatment could be interrupted for 28 months after anti-CD20 treatment. In patient two, thrombocytopenia followed a diagnosis of T1D 6 yr previously. Treatment with anti-CD20 led to normalization of thrombocytes, but no effect on T1D was observed. Concerning the origin of the boys' conditions, several primary immune dysregulation disorders were considered. Thrombocytopenia associated with T1D is unusual and could represent a new entity. The diabetes manifestation in patient one was probably triggered by corticosteroid treatment; regardless, anti-CD20 therapy appeared to be efficacious early in the course of T1D, but not long after the initial diagnosis of T1D, as shown for patient two.
Pubmed
Web of science
Création de la notice
26/03/2015 19:23
Dernière modification de la notice
03/03/2018 22:36
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