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    Autologous hematopoietic stem cell transplantation for Behçet's Disease: a retrospective survey of patients treated in Europe, on behalf of the Autoimmune Diseases Working Party of the European Society for Blood and Marrow Transplantation

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    Authors
    Puyade, M.
    Patel, Amit
    Lim, Y. J.
    Blank, N.
    Badoglio, M.
    Gualandi, F.
    Ma, D. D.
    Maximova, N.
    Greco, R.
    Alexander, T
    Snowden, J. A.
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    Affiliation
    CHU de Poitiers, Service de Médecine Interne et Maladies Infectieuses, Poitiers, France
    Issue Date
    2021
    
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    Abstract
    Background: Behçet's Disease (BD) is an autoimmune disease mostly presenting with recurrent oral and genital aphthosis, and uveitis. Patients are rarely refractory to immunosuppressive treatments. Autologous hematopoietic stem cell transplantation (aHSCT) is a standard of care in other autoimmune diseases. Some patients with BD have been treated with aHSCT based on compassionate use. Objectives: Evaluate the outcome of aHSCT in adult patients with BD treated in member centers of the European Society for Blood and Marrow Transplantation (EBMT). Methods: Adults who received aHSCT primarily for BD were identified retrospectively in the EBMT registry and/or in published literature. Data were extracted from either medical records of the patient or from publications. Results: Eight out of 9 cases reported to the registry and extracted data of 2 further patients from literature were analyzed. Four were female, median age at onset of BD was 24y (range 9-50). Median age at aHSCT was 32y (27-51). Patients had received median 4 (2-11) previous lines of therapy (89% corticosteroids, 50% methotrexate, anti-TNFα therapy or cyclophosphamide). All patients had active disease before mobilization. Conditioning regimen was heterogeneous. Median follow-up was 48 months (range 6-240). No treatment-related mortality was reported. This procedure induced complete remission (CR) in 80%, partial remission in 10% and lack of response in 10% of the patients. Relapse rate was 30% (2 relapses in patients in CR and 1 relapse in the patient in PR) with panuveitis (n=1), aphthosis (n=2) and arthralgia (n=1). Six patients were in CR. No late complications were reported. Conclusion: aHSCT has an acceptable safety profile and represents a feasible and relatively effective procedure in severe and conventional treatment-resistant cases of BD and has the potential to stabilize BD in patients with life-threatening involvements.
    Citation
    Puyade M, Patel A, Lim YJ, Blank N, Badoglio M, Gualandi F, et al. Autologous Hematopoietic Stem Cell Transplantation for Behçet’s Disease: A Retrospective Survey of Patients Treated in Europe, on Behalf of the Autoimmune Diseases Working Party of the European Society for Blood and Marrow Transplantation. Front Immunol. 2021 May 6;12. 
    Journal
    Frontiers in Immunology
    URI
    http://hdl.handle.net/10541/624095
    DOI
    10.3389/fimmu.2021.638709
    PubMed ID
    34025648
    Additional Links
    https://dx.doi.org/10.3389/fimmu.2021.638709
    Type
    Article
    Language
    en
    ae974a485f413a2113503eed53cd6c53
    10.3389/fimmu.2021.638709
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