Literature DB >> 34651299

Azacitidine for patients with Vacuoles, E1 Enzyme, X-linked, Autoinflammatory, Somatic syndrome (VEXAS) and myelodysplastic syndrome: data from the French VEXAS registry.

Thibault Comont1, Mael Heiblig2, Etienne Rivière3, Louis Terriou4, Julien Rossignol5, Didier Bouscary6, Virginie Rieu7, Guillaume Le Guenno7, Alexis Mathian8, Achille Aouba9, Julien Vinit10, Jeremie Dion1, Olivier Kosmider11, Benjamin Terrier12, Sophie Georgin-Lavialle13, Pierre Fenaux14, Arsène Mekinian15.   

Abstract

Azacitidine can be effective in myelodysplastic syndromes (MDS) associated with inflammatory/autoimmune diseases. Vacuoles, E1 Enzyme, X-linked, Autoinflammatory, Somatic syndrome (VEXAS) is a new monogenic autoinflammatory syndrome caused by somatic ubiquitin-like modifier-activating enzyme 1 (UBA1) mutation, often associated with MDS, whose treatment is difficult and not yet codified. Based on a French nationwide registry of 116 patients with VEXAS, we report the efficacy and safety of azacitidine treatment in 11 patients with VEXAS with MDS. Clinical response of VEXAS to azacitidine was achieved in five patients (46%), during 6, 8+, 12, 21, 27+ months respectively, suggesting that azacitidine can be effective in selected patients with VEXAS and associated MDS.
© 2021 British Society for Haematology and John Wiley & Sons Ltd.

Entities:  

Keywords:  VEXAS syndrome; azacitidine; myelodysplastic syndrome

Mesh:

Substances:

Year:  2021        PMID: 34651299     DOI: 10.1111/bjh.17893

Source DB:  PubMed          Journal:  Br J Haematol        ISSN: 0007-1048            Impact factor:   6.998


  11 in total

Review 1.  Update on VEXAS and role of allogeneic bone marrow transplant: Considerations on behalf of the Chronic Malignancies Working Party of the EBMT.

Authors:  Carmelo Gurnari; Donal P McLornan
Journal:  Bone Marrow Transplant       Date:  2022-08-08       Impact factor: 5.174

Review 2.  Vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic syndrome (VEXAS syndrome) with prominent supraglottic larynx involvement: a case-based review.

Authors:  Camila Andrea Guerrero-Bermúdez; Andrés Felipe Cardona-Cardona; Edwin Jesús Ariza-Parra; Juan Ignacio Arostegui; Anna Mensa-Vilaro; Jordi Yague; Gloria Vásquez; Carlos Horacio Muñoz-Vahos
Journal:  Clin Rheumatol       Date:  2022-08-20       Impact factor: 3.650

Review 3.  Mimickers of Large Vessel Giant Cell Arteritis.

Authors:  André Ramon; Hélène Greigert; Paul Ornetti; Bernard Bonnotte; Maxime Samson
Journal:  J Clin Med       Date:  2022-01-19       Impact factor: 4.241

4.  Distinction between clonal and paraclonal cutaneous involvements in VEXAS syndrome.

Authors:  Valentin Lacombe; Annaelle Beucher; Geoffrey Urbanski; Yannick Le Corre; Laurane Cottin; Anne Croué; Anne Bouvier
Journal:  Exp Hematol Oncol       Date:  2022-02-16

5.  Innovations in genomics for undiagnosed diseases: vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic (VEXAS) syndrome.

Authors:  Ryan J Stubbins; Hannah Cherniawsky; Luke Y C Chen; Thomas J Nevill
Journal:  CMAJ       Date:  2022-04-11       Impact factor: 8.262

Review 6.  VEXAS Syndrome: A Novelty in MDS Landscape.

Authors:  Marie Templé; Olivier Kosmider
Journal:  Diagnostics (Basel)       Date:  2022-06-29

7.  Case Report: Tocilizumab Treatment for VEXAS Syndrome With Relapsing Polychondritis: A Single-Center, 1-Year Longitudinal Observational Study In Japan.

Authors:  Yosuke Kunishita; Yohei Kirino; Naomi Tsuchida; Ayaka Maeda; Yuichiro Sato; Kaoru Takase-Minegishi; Ryusuke Yoshimi; Hideaki Nakajima
Journal:  Front Immunol       Date:  2022-06-13       Impact factor: 8.786

8. 

Authors:  Ryan J Stubbins; Hannah Cherniawsky; Luke Y C Chen; Thomas J Nevill
Journal:  CMAJ       Date:  2022-09-06       Impact factor: 16.859

9.  Comparison between idiopathic and VEXAS-relapsing polychondritis: analysis of a French case series of 95 patients.

Authors:  Mohamed-Yacine Khitri; Alexis F Guedon; Sophie Georgin-Lavialle; Benjamin Terrier; David Saadoun; Julie Seguier; Maelle le Besnerais; Claire De Moreuil; Guillaume Denis; Mathieu Gerfaud-Valentin; Jean Sebastien Allain; Alexandre Maria; Laurence Bouillet; Vincent Grobost; Joris Galland; Olivier Kosmider; Anael Dumont; Mathilde Devaux; Benjamin Subran; Jean Schmidt; Paola Marianetti-Guingel; Sylvain Audia; Sylvain Palat; Marielle Roux-Sauvat; Vincent Jachiet; Pierre Hirsch; Olivier Fain; Arsène Mekinian
Journal:  RMD Open       Date:  2022-07

Review 10.  Vasculitis associated with VEXAS syndrome: A literature review.

Authors:  Ryu Watanabe; Manami Kiji; Motomu Hashimoto
Journal:  Front Med (Lausanne)       Date:  2022-08-15
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