Real-life efficacy of immunotherapy for Sézary syndrome: a multicenter observational cohort study

Alizée Bozonnat; Marie Beylot-Barry; Olivier Dereure; Michel D'Incan; Gaëlle Quereux; Emmanuella Guenova; Marie Perier-Muzet; Stephane Dalle; Florent Grange; Manuelle-Anne Viguier; Caroline Ram-Wolff; Laurence Feldmeyer; Helmut Beltraminelli; Nathalie Bonnet; Florent Amatore; Eve Maubec; Nathalie Franck; Laurent Machet; François Chasset; Florence Brunet-Possenti; Jean-David Bouaziz; Maxime Battistella; Marie Donzel; Anne Pham-Ledard; Claudia Bejar; Hélène Moins-Teisserenc; Samia Mourah; Philippe Saiag; Ewa Hainaut; Catherine Michel; Guido Bens; Henri Adamski; François Aubin; Serge Boulinguez; Pascal Joly; Billal Tedbirt; Isabelle Templier; Laura Troin; Henri Montaudié; Saskia Ingen-Housz-Oro; Sarah Faiz; Laurent Mortier; Gabor Dobos; Martine Bagot; Matthieu Resche-Rigon; Claire Montlahuc; Arnaud Serret-Larmande; Adèle de Masson; Cutaneous Lymphomas French Study Group

doi:10.1016/j.eclinm.2024.102679

Real-life efficacy of immunotherapy for Sézary syndrome: a multicenter observational cohort study

Alizée Bozonnat
, Marie Beylot-Barry
, Olivier Dereure
, Michel D'Incan
, Gaëlle Quereux
, Emmanuella Guenova
, Marie Perier-Muzet
, Stephane Dalle
, Florent Grange
, Manuelle-Anne Viguier
, Caroline Ram-Wolff
, Laurence Feldmeyer
, Helmut Beltraminelli
, Nathalie Bonnet
, Florent Amatore
, Eve Maubec
, Nathalie Franck
, Laurent Machet
, François Chasset
, Florence Brunet-Possenti

Jean-David Bouaziz, Maxime Battistella, Marie Donzel, Anne Pham-Ledard, Claudia Bejar, Hélène Moins-Teisserenc, Samia Mourah, Philippe Saiag, Ewa Hainaut, Catherine Michel, Guido Bens, Henri Adamski, François Aubin, Serge Boulinguez, Pascal Joly, Billal Tedbirt, Isabelle Templier, Laura Troin, Henri Montaudié, Saskia Ingen-Housz-Oro, Sarah Faiz, Laurent Mortier, Gabor Dobos, Martine Bagot, Matthieu Resche-Rigon, Claire Montlahuc, Arnaud Serret-Larmande, Adèle de Masson, Cutaneous Lymphomas French Study Group

Research output: Contribution to journal › Article › peer-review

Abstract

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.

FUNDING: French Society of Dermatology, Swiss National Science Foundation (IZLIZ3_200253/1) and SKINTEGRITY.CH collaborative research program.

Original language	English
Article number	102679
Pages (from-to)	102679
Journal	EClinicalMedicine
Volume	73
DOIs	https://doi.org/10.1016/j.eclinm.2024.102679
Publication status	Published - Jul 2024
Externally published	Yes

Fields of science

302 Clinical Medicine

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

Access to Document

10.1016/j.eclinm.2024.102679

Cite this

Bozonnat, A., Beylot-Barry, M., Dereure, O., D'Incan, M., Quereux, G., Guenova, E., Perier-Muzet, M., Dalle, S., Grange, F., Viguier, M.-A., Ram-Wolff, C., Feldmeyer, L., Beltraminelli, H., Bonnet, N., Amatore, F., Maubec, E., Franck, N., Machet, L., Chasset, F., ... Cutaneous Lymphomas French Study Group (2024). Real-life efficacy of immunotherapy for Sézary syndrome: a multicenter observational cohort study. EClinicalMedicine, 73, 102679. Article 102679. https://doi.org/10.1016/j.eclinm.2024.102679

@article{ba76a032949241c5b35361ab5ab71942,

title = "Real-life efficacy of immunotherapy for S{\'e}zary syndrome: a multicenter observational cohort study",

abstract = "BACKGROUND: S{\'e}zary 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 S{\'e}zary syndrome, including treatment with mogamulizumab, in a real-life setting.METHODS: Data from patients with S{\'e}zary (ISCL/EORTC stage IV) and pre-S{\'e}zary (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-S{\'e}zary (9.7\% of 339), 296 S{\'e}zary 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 S{\'e}zary syndrome.FUNDING: French Society of Dermatology, Swiss National Science Foundation (IZLIZ3\_200253/1) and SKINTEGRITY.CH collaborative research program.",

author = "Aliz{\'e}e Bozonnat and Marie Beylot-Barry and Olivier Dereure and Michel D'Incan and Ga{\"e}lle Quereux and Emmanuella Guenova and Marie Perier-Muzet and Stephane Dalle and Florent Grange and Manuelle-Anne Viguier and Caroline Ram-Wolff and Laurence Feldmeyer and Helmut Beltraminelli and Nathalie Bonnet and Florent Amatore and Eve Maubec and Nathalie Franck and Laurent Machet and Fran{\c c}ois Chasset and Florence Brunet-Possenti and Jean-David Bouaziz and Maxime Battistella and Marie Donzel and Anne Pham-Ledard and Claudia Bejar and H{\'e}l{\`e}ne Moins-Teisserenc and Samia Mourah and Philippe Saiag and Ewa Hainaut and Catherine Michel and Guido Bens and Henri Adamski and Fran{\c c}ois Aubin and Serge Boulinguez and Pascal Joly and Billal Tedbirt and Isabelle Templier and Laura Troin and Henri Montaudi{\'e} and Saskia Ingen-Housz-Oro and Sarah Faiz and Laurent Mortier and Gabor Dobos and Martine Bagot and Matthieu Resche-Rigon and Claire Montlahuc and Arnaud Serret-Larmande and \{de Masson\}, Ad{\`e}le and \{Cutaneous Lymphomas French Study Group\}",

note = "{\textcopyright} 2024 The Authors.",

year = "2024",

month = jul,

doi = "10.1016/j.eclinm.2024.102679",

language = "English",

volume = "73",

pages = "102679",

journal = "EClinicalMedicine",

issn = "2589-5370",

}

Bozonnat, A, Beylot-Barry, M, Dereure, O, D'Incan, M, Quereux, G, Guenova, E, Perier-Muzet, M, Dalle, S, Grange, F, Viguier, M-A, Ram-Wolff, C, Feldmeyer, L, Beltraminelli, H, Bonnet, N, Amatore, F, Maubec, E, Franck, N, Machet, L, Chasset, F, Brunet-Possenti, F, Bouaziz, J-D, Battistella, M, Donzel, M, Pham-Ledard, A, Bejar, C, Moins-Teisserenc, H, Mourah, S, Saiag, P, Hainaut, E, Michel, C, Bens, G, Adamski, H, Aubin, F, Boulinguez, S, Joly, P, Tedbirt, B, Templier, I, Troin, L, Montaudié, H, Ingen-Housz-Oro, S, Faiz, S, Mortier, L, Dobos, G, Bagot, M, Resche-Rigon, M, Montlahuc, C, Serret-Larmande, A, de Masson, A & Cutaneous Lymphomas French Study Group 2024, 'Real-life efficacy of immunotherapy for Sézary syndrome: a multicenter observational cohort study', EClinicalMedicine, vol. 73, 102679, pp. 102679. https://doi.org/10.1016/j.eclinm.2024.102679

TY - JOUR

T1 - Real-life efficacy of immunotherapy for Sézary syndrome

T2 - a multicenter observational cohort study

AU - Bozonnat, Alizée

AU - Beylot-Barry, Marie

AU - Dereure, Olivier

AU - D'Incan, Michel

AU - Quereux, Gaëlle

AU - Guenova, Emmanuella

AU - Perier-Muzet, Marie

AU - Dalle, Stephane

AU - Grange, Florent

AU - Viguier, Manuelle-Anne

AU - Ram-Wolff, Caroline

AU - Feldmeyer, Laurence

AU - Beltraminelli, Helmut

AU - Bonnet, Nathalie

AU - Amatore, Florent

AU - Maubec, Eve

AU - Franck, Nathalie

AU - Machet, Laurent

AU - Chasset, François

AU - Brunet-Possenti, Florence

AU - Bouaziz, Jean-David

AU - Battistella, Maxime

AU - Donzel, Marie

AU - Pham-Ledard, Anne

AU - Bejar, Claudia

AU - Moins-Teisserenc, Hélène

AU - Mourah, Samia

AU - Saiag, Philippe

AU - Hainaut, Ewa

AU - Michel, Catherine

AU - Bens, Guido

AU - Adamski, Henri

AU - Aubin, François

AU - Boulinguez, Serge

AU - Joly, Pascal

AU - Tedbirt, Billal

AU - Templier, Isabelle

AU - Troin, Laura

AU - Montaudié, Henri

AU - Ingen-Housz-Oro, Saskia

AU - Faiz, Sarah

AU - Mortier, Laurent

AU - Dobos, Gabor

AU - Bagot, Martine

AU - Resche-Rigon, Matthieu

AU - Montlahuc, Claire

AU - Serret-Larmande, Arnaud

AU - de Masson, Adèle

AU - Cutaneous Lymphomas French Study Group

PY - 2024/7

Y1 - 2024/7

N2 - 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.FUNDING: French Society of Dermatology, Swiss National Science Foundation (IZLIZ3_200253/1) and SKINTEGRITY.CH collaborative research program.

AB - 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.FUNDING: French Society of Dermatology, Swiss National Science Foundation (IZLIZ3_200253/1) and SKINTEGRITY.CH collaborative research program.

UR - https://www.scopus.com/pages/publications/85196398449

U2 - 10.1016/j.eclinm.2024.102679

DO - 10.1016/j.eclinm.2024.102679

M3 - Article

C2 - 39007062

SN - 2589-5370

VL - 73

SP - 102679

JO - EClinicalMedicine

JF - EClinicalMedicine

M1 - 102679