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Dupilumab for relapsing or refractory sinonasal and/or asthma manifestations in eosinophilic granulomatosis with polyangiitis : a European retrospective study

Molina, Berengere (författare)
Hosp Cochin, Dept Internal Med, Paris, France.
Padoan, Roberto (författare)
Univ Padua, Dept Med DIMED, Div Rheumatol, Padua, Italy.
Urban, Maria Letizia (författare)
Univ Florence, Dept Expt & Clin Med, Florence, Italy.
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Novikov, Pavel (författare)
IM Sechenov First Moscow State Med Univ, Tareev Clin Internal Dis, Moscow, Russia.
Caminati, Marco (författare)
Univ Verona, Dept Med Asthma Allergy & Clin Immunol, Verona, Italy.
Taillé, Camille (författare)
Nord Univ Paris Cite, Bichat Hosp, Dept Resp Dis, APHP, Paris, France.
Néel, Antoine (författare)
Nantes Univ Hosp, Dept Internal Med, Nantes, France.
Bouillet, Laurence (författare)
Grenoble Alpes Univ Hosp, T Reg Unit, CNRS, UMR 5525,5,VetAgro Sup, Grenoble, France.;Grenoble Univ Hosp, Internal Med Dept, Grenoble, France.
Fraticelli, Paolo (författare)
Univ Hosp Osped Riuniti, Internal Med Dept, Ancona, Italy.
Schleinitz, Nicolas (författare)
Aix Marseille Univ, Timone Hosp AP HM, Dept Internal Med, Marseille, France.
Christides, Christine (författare)
Avignon Hosp, Dept Internal Med, Avignon, France.
Moi, Laura (författare)
Valais Hosp, Inst Cent Hop, Immunol & Allergol, Sion, Switzerland.
Godeau, Bertrand (författare)
Henri Mondor Univ Hosp, Dept Internal Med, Creteil, France.
Knight, Ann (författare)
Uppsala universitet,Reumatologi
Schroeder, Jan Walter (författare)
ASST Grande Osped Metropolitano Niguarda, Unit Allergol & Immunol, Milan, Italy.
Marchand-Adam, Sylvain (författare)
Reg Univ Hosp Ctr, Dept Resp Med, Tours, France.
Gil, Helder (författare)
Besancon Univ Hosp, Dept Internal Med, Besancon, France.
Cottin, Vincent (författare)
Louis Pradel Hosp, Coordinating Reference Ctr Rare Pulm Dis, Dept Resp Med, Bron, France.
Durel, Cécile-Audrey (författare)
Univ Hosp Edouard Herriot, Dept Internal Med, HCL, Lyon, France.
Gelain, Elena (författare)
Meyer Childrens Hosp, Nephrol & Dialysis Unit, Florence, Italy.
Lerais, Boris (författare)
Brest Univ Hosp, Dept Internal Med, Brest, France.
Ruivard, Marc (författare)
Univ Hosp Ctr, Internal Med, Clermont Ferrand, France.
Groh, Matthieu (författare)
Foch Hosp, Natl Referral Ctr Hypereosinophil Syndrome CEREO, Dept Internal Med, Suresnes, France.
Samson, Maxime (författare)
Univ Hosp Ctr, Dept Internal Med & Clin Immunol, Dijon, France.;Univ Bourgogne Franche Comte, INSERM, EFS BFC, UMR1098,RIGHT Interact Greffon Hote Tumeur Ingenie, Dijon, France.
Moroni, Luca (författare)
Ist Sci San Raffaele, Unit Immunol Rheumatol Allergy & Rare Dis UnIRAR, Milan, Italy.
Thiel, Jens (författare)
Univ Freiburg, Med Ctr, Dept Rheumatol & Clin Immunol, Freiburg, Germany.;Med Univ, Dept Internal Med, Div Rheumatol & Clin Immunol, Graz, Austria.
Kernder, Anna (författare)
Heinrich Heine Univ Dusseldorf, Med Fac, Dept Rheumatol & Hiller Res Unit Rheumatol, Dusseldorf, Germany.
Cohen Tervaert, Jan Willem (författare)
Univ Alberta, Dept Med, Edmonton, AB, Canada.;Maastricht Univ, Sch Mental Hlth & Neurosci, Maastricht, Netherlands.
Costanzo, Giulia (författare)
Univ Cagliari, Dept Med Sci & Publ Hlth, Monserrato, Italy.
Folci, Marco (författare)
Fdn Poliambulanza Ist Osped, Brescia, Italy.
Rizzello, Sonia (författare)
Careggi Univ Hosp, Severe Asthma Unit, Florence, Italy.
Cohen, Pascal (författare)
Cochin Hosp, Dept Internal Med, Paris, France.
Emmi, Giacomo (författare)
Univ Florence, Dept Expt & Clin Med, Florence, Italy.;Monash Univ, Ctr Inflammatory Dis, Monash Med Ctr, Dept Med, Clayton, Vic, Australia.
Terrier, Benjamin (författare)
Hosp Cochin, Dept Internal Med, F-75014 Paris, Ile De France, France.;Hosp Cochin, Dept Internal Med, Paris, France.
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Hosp Cochin, Dept Internal Med, Paris, France Univ Padua, Dept Med DIMED, Div Rheumatol, Padua, Italy. (creator_code:org_t)
BMJ Publishing Group Ltd, 2023
2023
Engelska.
Ingår i: Annals of the Rheumatic Diseases. - : BMJ Publishing Group Ltd. - 0003-4967 .- 1468-2060. ; 82:12, s. 1587-1593
Relaterad länk:
https://urn.kb.se/re...
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https://doi.org/10.1...
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  • Tidskriftsartikel (refereegranskat)
Abstract Ämnesord
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  • Background Eosinophilic granulomatosis with polyangiitis (EGPA) is often associated with glucocorticoid-dependent asthma and/or ear, nose and throat (ENT) manifestations. When immunosuppressants and/or mepolizumab are ineffective, dupilumab could be an option. We describe the safety and efficacy of off-label use of dupilumab in relapsing and/or refractory EGPA.Patients and methods We conducted an observational multicentre study of EGPA patients treated with dupilumab. Complete response was defined by Birmingham Vasculitis Activity Score (BVAS)=0 and prednisone dose ≤4 mg/day, and partial response by BVAS=0 and prednisone dose >4 mg/day. Eosinophilia was defined as an eosinophil count >500/mm3.Results Fifty-one patients were included. The primary indication for dupilumab was disabling ENT symptoms in 92%. After a median follow-up of 13.1 months, 18 patients (35%) reported adverse events (AEs), including two serious AEs. Eosinophilia was reported in 34 patients (67%), with a peak of 2195/mm3 (IQR 1268–4501) occurring at 13 weeks (IQR 4–36) and was associated with relapse in 41%. Twenty-one patients (41%) achieved a complete response and 12 (24%) a partial response. Sixteen (31%) patients experienced an EGPA relapse while on dupilumab, which was associated with blood eosinophilia in 14/16 (88%) patients. The median eosinophil count at the start of dupilumab was significantly lower in relapsers than in non-relapsers, as was the median time between stopping anti-IL-5/IL-5R and switching to dupilumab.Conclusion These results suggest that dupilumab may be effective in treating patients with EGPA-related ENT manifestations. However, EGPA flares occurred in one-third of patients and were preceded by eosinophilia in 88%, suggesting that caution is required.

Ämnesord

MEDICIN OCH HÄLSOVETENSKAP  -- Klinisk medicin -- Reumatologi och inflammation (hsv//swe)
MEDICAL AND HEALTH SCIENCES  -- Clinical Medicine -- Rheumatology and Autoimmunity (hsv//eng)
MEDICIN OCH HÄLSOVETENSKAP  -- Medicinska och farmaceutiska grundvetenskaper -- Immunologi inom det medicinska området (hsv//swe)
MEDICAL AND HEALTH SCIENCES  -- Basic Medicine -- Immunology in the medical area (hsv//eng)

Nyckelord

Systemic vasculitis
Immune System Diseases
Biological Therapy
Autoimmune Diseases
Therapeutics

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