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

Article Article scientifique Rapport de cas Diabetes and immune thrombocytopenic purpura: a new association with good response to anti-CD20 therapy eng 2015 author von Laer Tschudin Letizia author Schwitzgebel Luscher Valérie 133261 author von Scheven-Gête Annette author Blouin Jean-Louis 513663 author Hofer Michael 31804 author Hauschild Michael author Ansari Djaberi Marc Georges 61572 author Stoppa Sophie 107968 author Phan-Hug Franziska 138-145 10.1111/pedi.12128 24552605 1399-543X 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. Dewey 576.5 Dewey 618 135 Département de médecine génétique et développement 21D 58 Département de pédiatrie, gynécologie et obstétrique 223 false 144 Diabète et métabolisme 178 564 Oncologie et hématologie pédiatrique 907 Pediatric diabetes 16 2 53fe7201-a6dd-43fe-8fcc-95cf34362835 Article (Published version) Restricted 138-145.pdf 939951 application/pdf restrictedAccess Faculté de médecine / Section de médecine fondamentale / Département de médecine génétique et développement Faculté de médecine / Section de médecine clinique / Département de pédiatrie, gynécologie et obstétrique

Title	Published in	Year	Views	Downloads

Diabetes and immune thrombocytopenic purpura: a new association with good response to anti-CD20 therapy	Pediatric diabetes	2015	1,139	1
État inflammatoire et prothrombogene chez les enfants obèses d'origine européenne		2011	973	763

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