Real-life efficacy of immunotherapy for Sézary syndrome: a multicenter observational cohort study
Alizée Bozonnat
(1, 2, 3)
,
Marie Beylot-Barry
(4, 5)
,
Olivier Dereure
(6)
,
Michel d'Incan
(7)
,
Gaëlle Quereux
(8, 9, 10)
,
Emmanuella Guenova
(11, 12)
,
Marie Perier-Muzet
(13)
,
Stephane Dalle
(13)
,
Florent Grange
(14)
,
Manuelle-Anne Viguier
(15, 16)
,
Caroline Ram-Wolff
(3)
,
Laurence Feldmeyer
(17, 18)
,
Helmut Beltraminelli
(17, 18)
,
Nathalie Bonnet
(19)
,
Florent Amatore
(19)
,
Eve Maubec
(20)
,
Nathalie Franck
(21)
,
Laurent Machet
(22)
,
François Chasset
(23)
,
Florence Brunet-Possenti
(24)
,
Jean-David Bouaziz
(3, 2, 1)
,
Maxime Battistella
(1, 2, 25)
,
Marie Donzel
(13)
,
Anne Pham-Ledard
(26, 5)
,
Claudia Bejar
(20)
,
Hélène Moins-Teisserenc
(2, 25)
,
Samia Mourah
(2, 1, 25)
,
Philippe Saiag
(27, 28, 29)
,
Ewa Hainaut
(30)
,
Catherine Michel
(31)
,
Guido Bens
(32)
,
Henri Adamski
(33)
,
François Aubin
(34)
,
Serge Boulinguez
(35)
,
Pascal Joly
(36)
,
Billal Tedbirt
(36)
,
Isabelle Templier
(37)
,
Laura Troin
(38)
,
Henri Montaudié
(38)
,
Saskia Ingen-Housz-Oro
(39)
,
Sarah Faiz
(40)
,
Laurent Mortier
(40)
,
Gabor Dobos
(41)
,
Martine Bagot
(3, 2, 1)
,
Matthieu Resche-Rigon
(1, 2, 42)
,
Claire Montlahuc
(42)
,
Arnaud Serret-Larmande
(42, 2, 1)
,
Adèle de Masson
(2, 1, 3)
1
HIPI (UMR_S_976 / U976) -
Immunologie humaine, physiopathologie & immunothérapie
2 UPCité - Université Paris Cité
3 Service de Dermatologie [AP-HP Hôpital Saint-Louis]
4 CHU Bordeaux - Centre Hospitalier Universitaire de Bordeaux
5 Inserm U1312 - BRIC - BoRdeaux Institute in onCology
6 CHRU Montpellier - Centre Hospitalier Régional Universitaire [Montpellier]
7 Service Dermatologie et Oncologie Cutanée [CHU Clermont-Ferrand]
8 INCIT - Immunology and New Concepts in ImmunoTherapy
9 CHU Nantes - Centre Hospitalier Universitaire de Nantes = Nantes University Hospital
10 CIC Nantes - Centre d’Investigation Clinique de Nantes
11 UNIL - Université de Lausanne = University of Lausanne
12 CHUV - Centre Hospitalier Universitaire Vaudois = Lausanne University Hospital [Lausanne]
13 HCL - Hospices Civils de Lyon
14 Centre hospitalier de Valence
15 URCA - Université de Reims Champagne-Ardenne
16 IRMAIC - Immuno-Régulation dans les Maladies Auto-Immunes Inflammatoires et le Cancer - EA 7509
17 UNIBE - Universität Bern = University of Bern = Université de Berne
18 Inselspital - Bern University Hospital [Berne]
19 CHU Marseille
20 Service de dermatologie [Avicenne]
21 Service de Dermatologie [CHU Cochin]
22 CHU Trousseau [Tours]
23 CHU Tenon [AP-HP]
24 AP-HP - Hôpital Bichat - Claude Bernard [Paris]
25 AP-HP - Hopital Saint-Louis [AP-HP]
26 Service de dermatologie [Bordeaux]
27 Hôpital Ambroise Paré [AP-HP]
28 UVSQ - Université de Versailles Saint-Quentin-en-Yvelines
29 Université Paris-Saclay
30 CHU de Poitiers [La Milétrie] - Centre hospitalier universitaire de Poitiers = Poitiers University Hospital
31 CH E.Muller Mulhouse - Centre Hospitalier Emile Muller [Mulhouse]
32 CHRO - Centre Hospitalier Régional d'Orléans
33 Centre Hospitalier Universitaire de Rennes [CHU Rennes] = Rennes University Hospital [Pontchaillou]
34 CHRU Besançon - Centre Hospitalier Régional Universitaire de Besançon
35 Service Dermatologie [CHU Toulouse]
36 Service de dermatologie [Rouen]
37 CHUGA - Centre Hospitalier Universitaire [CHU Grenoble]
38 CHU Nice - Centre Hospitalier Universitaire de Nice
39 Hôpital Henri Mondor
40 CHRU Lille - Centre Hospitalier Régional Universitaire [CHU Lille]
41 Charité - UniversitätsMedizin = Berlin University Medicine
42 Service de biostatistiques et information médicale [Saint-Louis]
2 UPCité - Université Paris Cité
3 Service de Dermatologie [AP-HP Hôpital Saint-Louis]
4 CHU Bordeaux - Centre Hospitalier Universitaire de Bordeaux
5 Inserm U1312 - BRIC - BoRdeaux Institute in onCology
6 CHRU Montpellier - Centre Hospitalier Régional Universitaire [Montpellier]
7 Service Dermatologie et Oncologie Cutanée [CHU Clermont-Ferrand]
8 INCIT - Immunology and New Concepts in ImmunoTherapy
9 CHU Nantes - Centre Hospitalier Universitaire de Nantes = Nantes University Hospital
10 CIC Nantes - Centre d’Investigation Clinique de Nantes
11 UNIL - Université de Lausanne = University of Lausanne
12 CHUV - Centre Hospitalier Universitaire Vaudois = Lausanne University Hospital [Lausanne]
13 HCL - Hospices Civils de Lyon
14 Centre hospitalier de Valence
15 URCA - Université de Reims Champagne-Ardenne
16 IRMAIC - Immuno-Régulation dans les Maladies Auto-Immunes Inflammatoires et le Cancer - EA 7509
17 UNIBE - Universität Bern = University of Bern = Université de Berne
18 Inselspital - Bern University Hospital [Berne]
19 CHU Marseille
20 Service de dermatologie [Avicenne]
21 Service de Dermatologie [CHU Cochin]
22 CHU Trousseau [Tours]
23 CHU Tenon [AP-HP]
24 AP-HP - Hôpital Bichat - Claude Bernard [Paris]
25 AP-HP - Hopital Saint-Louis [AP-HP]
26 Service de dermatologie [Bordeaux]
27 Hôpital Ambroise Paré [AP-HP]
28 UVSQ - Université de Versailles Saint-Quentin-en-Yvelines
29 Université Paris-Saclay
30 CHU de Poitiers [La Milétrie] - Centre hospitalier universitaire de Poitiers = Poitiers University Hospital
31 CH E.Muller Mulhouse - Centre Hospitalier Emile Muller [Mulhouse]
32 CHRO - Centre Hospitalier Régional d'Orléans
33 Centre Hospitalier Universitaire de Rennes [CHU Rennes] = Rennes University Hospital [Pontchaillou]
34 CHRU Besançon - Centre Hospitalier Régional Universitaire de Besançon
35 Service Dermatologie [CHU Toulouse]
36 Service de dermatologie [Rouen]
37 CHUGA - Centre Hospitalier Universitaire [CHU Grenoble]
38 CHU Nice - Centre Hospitalier Universitaire de Nice
39 Hôpital Henri Mondor
40 CHRU Lille - Centre Hospitalier Régional Universitaire [CHU Lille]
41 Charité - UniversitätsMedizin = Berlin University Medicine
42 Service de biostatistiques et information médicale [Saint-Louis]
Marie Donzel
- Fonction : Auteur
- PersonId : 1430936
- ORCID : 0000-0002-5797-5744
Anne Pham-Ledard
- Fonction : Auteur
Adèle de Masson
- Fonction : Auteur
- PersonId : 798790
- ORCID : 0000-0001-7828-6211
- IdRef : 223880744
Résumé
Background: Sézary syndrome is an extremely rare and fatal cutaneous T-cell lymphoma (CTCL). Mogamulizumab, an anti-CCR4 monoclonal antibody, has recently been associated with increased progression-free survival in a randomized clinical trial in CTCL. We aimed to evaluate OS and prognostic factors in Sézary syndrome, including treatment with mogamulizumab, in a real-life setting.
Methods: Data from patients with Sézary (ISCL/EORTC stage IV) and pre-Sézary (stage IIIB) syndrome diagnosed from 2000 to 2020 were obtained from 24 centers in Europe. Age, disease stage, plasma lactate dehydrogenases levels, blood eosinophilia at diagnosis, large-cell transformation and treatment received were analyzed in a multivariable Cox proportional hazard ratio model. This study has been registered in ClinicalTrials (SURPASSe01 study: NCT05206045).
Findings: Three hundred and thirty-nine patients were included (58% men, median age at diagnosis of 70 years, Q1-Q3, 61-79): 33 pre-Sézary (9.7% of 339), 296 Sézary syndrome (87.3%), of whom 10 (2.9%) had large-cell transformation. One hundred and ten patients received mogamulizumab. Median follow-up was 58 months (95% confidence interval [CI], 53-68). OS was 46.5% (95% CI, 40.6%-53.3%) at 5 years. Multivariable analysis showed that age ≥ 80 versus <50 (HR: 4.9, 95% CI, 2.1-11.2, p = 0.001), and large-cell transformation (HR: 2.8, 95% CI, 1.6-5.1, p = 0.001) were independent and significant factors associated with reduced OS. Mogamulizumab treatment was significantly associated with decreased mortality (HR: 0.34, 95% CI, 0.15-0.80, p = 0.013).
Interpretation: Treatment with mogamulizumab was significantly and independently associated with decreased mortality in Sézary syndrome.
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