Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study
Background What baseline predictors would be involved in mortality in people with primary Sjögren syndrome (SjS) remains uncertain. This study aimed to investigate the baseline characteristics collected at the time of diagnosis of SjS associated with mortality and to identify mortality risk factors...
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| Format: | article |
| Langue: | anglais |
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2023
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| Accès en ligne: | https://hdl.handle.net/20.500.12008/52138 |
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| _version_ | 1868890149571526656 |
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| author | Brito-Zerón, Pilar |
| author2 | Flores-Chávez, Alejandra Horváth, Ildiko Fanny Rasmussen, Astrid Xiaomei, Li Olsson, Peter Vissink, Arjan J. Priori, Roberta Armaǧan, Berkan Hernández-Molina, Gabriela Consani, Sandra |
| author2_role | author author author author author author author author author author |
| author_browse | Armaǧan, Berkan Brito-Zerón, Pilar Consani, Sandra Flores-Chávez, Alejandra Hernández-Molina, Gabriela Horváth, Ildiko Fanny Olsson, Peter Priori, Roberta Rasmussen, Astrid Vissink, Arjan J. Xiaomei, Li |
| author_facet | Brito-Zerón, Pilar Flores-Chávez, Alejandra Horváth, Ildiko Fanny Rasmussen, Astrid Xiaomei, Li Olsson, Peter Vissink, Arjan J. Priori, Roberta Armaǧan, Berkan Hernández-Molina, Gabriela Consani, Sandra |
| author_role | author |
| collection | COLIBRI |
| dc.contributor.none.fl_str_mv | Brito-Zerón Pilar Flores-Chávez Alejandra Horváth Ildiko Fanny Rasmussen Astrid Xiaomei Li Olsson Peter Vissink Arjan J. Priori Roberta Armaǧan Berkan Hernández-Molina Gabriela Consani Sandra, Universidad de la República (Uruguay). Facultad de Medicina |
| dc.creator.none.fl_str_mv | Brito-Zerón, Pilar Flores-Chávez, Alejandra Horváth, Ildiko Fanny Rasmussen, Astrid Xiaomei, Li Olsson, Peter Vissink, Arjan J. Priori, Roberta Armaǧan, Berkan Hernández-Molina, Gabriela Consani, Sandra |
| dc.date.none.fl_str_mv | 2023 2025-10-17T17:08:56Z 2025-10-17T17:08:56Z |
| dc.format.none.fl_str_mv | 16 p. application/pdf |
| dc.identifier.none.fl_str_mv | Brito-Zerón P, Flores-Chávez A, Horváth I y otros. Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study. eClinicalMedicine [en línea]. 2023;61. 16 p. 2589-5370 https://hdl.handle.net/20.500.12008/52138 10.1016/j.eclinm.2023.102062 |
| dc.language.none.fl_str_mv | en eng |
| dc.publisher.none.fl_str_mv | Elsevier |
| dc.relation.none.fl_str_mv | eClinicalMedicine. 2023;61 |
| dc.rights.none.fl_str_mv | info:eu-repo/semantics/openAccess Licencia Creative Commons Atribución - No Comercial - Sin Derivadas (CC - By-NC-ND 4.0) |
| dc.source.none.fl_str_mv | reponame:COLIBRI instname:Universidad de la República instacron:Universidad de la República |
| dc.subject.none.fl_str_mv | Cardiovascular Infection Lymphoma Mortality Sjögren syndrome Systemic disease SÍNDROME DE SJÖGREN METABOLISMO BASAL MORTALIDAD LINFOMA INFECCIONES |
| dc.title.none.fl_str_mv | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study |
| dc.type.none.fl_str_mv | Artículo info:eu-repo/semantics/article info:eu-repo/semantics/publishedVersion |
| description | Background What baseline predictors would be involved in mortality in people with primary Sjögren syndrome (SjS) remains uncertain. This study aimed to investigate the baseline characteristics collected at the time of diagnosis of SjS associated with mortality and to identify mortality risk factors for all-cause death and deaths related to systemic SjS activity measured by the ESSDAI score. Methods In this international, real-world, retrospective, cohort study, we retrospectively collected data from 27 countries on mortality and causes of death from the Big Data Sjögren Registry. Inclusion criteria consisted of fulfilling 2002/2016 SjS classification criteria, and exclusion criteria included chronic HCV/HIV infections and associated systemic autoimmune diseases. A statistical approach based on a directed acyclic graph was used, with all-cause and Sjögren-related mortality as primary endpoints. The key determinants that defined the disease phenotype at diagnosis (glandular, systemic, and immunological) were analysed as independent variables. Findings Between January 1st, 2014 and December 31, 2023, data from 11,372 patients with primary SjS (93.5% women, 78.4% classified as White, mean age at diagnosis of 51.1 years) included in the Registry were analysed. 876 (7.7%) deaths were recorded after a mean follow-up of 8.6 years (SD 7.12). Univariate analysis of prognostic factors for all-cause death identified eight Sjögren-related variables (ocular and oral tests, salivary biopsy, ESSDAI, ANA, anti-Ro, anti-La, and cryoglobulins). The multivariate CPH model adjusted for these variables and the epidemiological features showed that DAS-ESSDAI (high vs no high: HR = 1.68; 95% CI, 1.27–2.22) and cryoglobulins (positive vs negative: HR = 1.72; 95% CI, 1.22–2.42) were independent predictors of all-cause death. Of the 640 deaths with available information detailing the specific cause of death, 14% were due to systemic SjS. Univariate analysis of prognostic factors for Sjögren-cause death identified five Sjögren-related variables (oral tests, clinESSDAI, DAS-ESSDAI, ANA, and cryoglobulins). The multivariate competing risks CPH model adjusted for these variables and the epidemiological features showed that oral tests (abnormal vs normal results: HR = 1.38; 95% CI, 1.01–1.87), DAS-ESSDAI (high vs no high: HR = 1.55; 95% CI, 1.22–1.96) and cryoglobulins (positive vs negative: HR = 1.52; 95% CI, 1.16–2) were independent predictors of SjS-related death. Interpretation The key mortality risk factors at the time of SjS diagnosis were positive cryoglobulins and a high systemic activity scored using the ESSDAI, conferring a 2-times increased risk of all-cause and SjS-related death. ESSDAI measurement and cryoglobulin testing should be considered mandatory when an individual is diagnosed with SjS. |
| eu_rights_str_mv | openAccess |
| format | article |
| id | anni_a08c434aa1d1dd176cda2d683bb57c2d |
| identifier_str_mv | Brito-Zerón P, Flores-Chávez A, Horváth I y otros. Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study. eClinicalMedicine [en línea]. 2023;61. 16 p. 2589-5370 10.1016/j.eclinm.2023.102062 |
| instacron_str | Universidad de la República |
| institution | Universidad de la República |
| instname_str | Universidad de la República |
| language | eng |
| language_invalid_str_mv | en |
| network_acronym_str | anni |
| network_name_str | oai-lr-anni |
| oai_identifier_str | oai:colibri.udelar.edu.uy:20.500.12008/52138 |
| publishDate | 2023 |
| publishDateSort | 2023 |
| publisher.none.fl_str_mv | Elsevier |
| reponame_str | COLIBRI |
| repository.mail.fl_str_mv | |
| repository.name.fl_str_mv | |
| repository_id_str | |
| rights_invalid_str_mv | Licencia Creative Commons Atribución - No Comercial - Sin Derivadas (CC - By-NC-ND 4.0) |
| spelling | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort studyBrito-Zerón, PilarFlores-Chávez, AlejandraHorváth, Ildiko FannyRasmussen, AstridXiaomei, LiOlsson, PeterVissink, Arjan J.Priori, RobertaArmaǧan, BerkanHernández-Molina, GabrielaConsani, SandraCardiovascularInfectionLymphomaMortalitySjögren syndromeSystemic diseaseSÍNDROME DE SJÖGRENMETABOLISMO BASALMORTALIDADLINFOMAINFECCIONESBackground What baseline predictors would be involved in mortality in people with primary Sjögren syndrome (SjS) remains uncertain. This study aimed to investigate the baseline characteristics collected at the time of diagnosis of SjS associated with mortality and to identify mortality risk factors for all-cause death and deaths related to systemic SjS activity measured by the ESSDAI score. Methods In this international, real-world, retrospective, cohort study, we retrospectively collected data from 27 countries on mortality and causes of death from the Big Data Sjögren Registry. Inclusion criteria consisted of fulfilling 2002/2016 SjS classification criteria, and exclusion criteria included chronic HCV/HIV infections and associated systemic autoimmune diseases. A statistical approach based on a directed acyclic graph was used, with all-cause and Sjögren-related mortality as primary endpoints. The key determinants that defined the disease phenotype at diagnosis (glandular, systemic, and immunological) were analysed as independent variables. Findings Between January 1st, 2014 and December 31, 2023, data from 11,372 patients with primary SjS (93.5% women, 78.4% classified as White, mean age at diagnosis of 51.1 years) included in the Registry were analysed. 876 (7.7%) deaths were recorded after a mean follow-up of 8.6 years (SD 7.12). Univariate analysis of prognostic factors for all-cause death identified eight Sjögren-related variables (ocular and oral tests, salivary biopsy, ESSDAI, ANA, anti-Ro, anti-La, and cryoglobulins). The multivariate CPH model adjusted for these variables and the epidemiological features showed that DAS-ESSDAI (high vs no high: HR = 1.68; 95% CI, 1.27–2.22) and cryoglobulins (positive vs negative: HR = 1.72; 95% CI, 1.22–2.42) were independent predictors of all-cause death. Of the 640 deaths with available information detailing the specific cause of death, 14% were due to systemic SjS. Univariate analysis of prognostic factors for Sjögren-cause death identified five Sjögren-related variables (oral tests, clinESSDAI, DAS-ESSDAI, ANA, and cryoglobulins). The multivariate competing risks CPH model adjusted for these variables and the epidemiological features showed that oral tests (abnormal vs normal results: HR = 1.38; 95% CI, 1.01–1.87), DAS-ESSDAI (high vs no high: HR = 1.55; 95% CI, 1.22–1.96) and cryoglobulins (positive vs negative: HR = 1.52; 95% CI, 1.16–2) were independent predictors of SjS-related death. Interpretation The key mortality risk factors at the time of SjS diagnosis were positive cryoglobulins and a high systemic activity scored using the ESSDAI, conferring a 2-times increased risk of all-cause and SjS-related death. ESSDAI measurement and cryoglobulin testing should be considered mandatory when an individual is diagnosed with SjS.ElsevierBrito-Zerón PilarFlores-Chávez AlejandraHorváth Ildiko FannyRasmussen AstridXiaomei LiOlsson PeterVissink Arjan J.Priori RobertaArmaǧan BerkanHernández-Molina GabrielaConsani Sandra, Universidad de la República (Uruguay). Facultad de Medicina2025-10-17T17:08:56Z2025-10-17T17:08:56Z2023Artículoinfo:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersion16 p.application/pdfBrito-Zerón P, Flores-Chávez A, Horváth I y otros. Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study. eClinicalMedicine [en línea]. 2023;61. 16 p.2589-5370https://hdl.handle.net/20.500.12008/5213810.1016/j.eclinm.2023.102062reponame:COLIBRIinstname:Universidad de la Repúblicainstacron:Universidad de la RepúblicaenengeClinicalMedicine. 2023;61Las obras depositadas en el Repositorio se rigen por la Ordenanza de los Derechos de la Propiedad Intelectual de la Universidad de la República.(Res. Nº 91 de C.D.C. de 8/III/1994 – D.O. 7/IV/1994) y por la Ordenanza del Repositorio Abierto de la Universidad de la República (Res. Nº 16 de C.D.C. de 07/10/2014)info:eu-repo/semantics/openAccessLicencia Creative Commons Atribución - No Comercial - Sin Derivadas (CC - By-NC-ND 4.0)oai:colibri.udelar.edu.uy:20.500.12008/521382026-04-14T10:28:20Z |
| spellingShingle | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study Brito-Zerón, Pilar Cardiovascular Infection Lymphoma Mortality Sjögren syndrome Systemic disease SÍNDROME DE SJÖGREN METABOLISMO BASAL MORTALIDAD LINFOMA INFECCIONES |
| status_str | publishedVersion |
| title | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study |
| title_full | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study |
| title_fullStr | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study |
| title_full_unstemmed | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study |
| title_short | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study |
| title_sort | Mortality risk factors in primary Sjögren syndrome: a real-world, retrospective, cohort study |
| topic | Cardiovascular Infection Lymphoma Mortality Sjögren syndrome Systemic disease SÍNDROME DE SJÖGREN METABOLISMO BASAL MORTALIDAD LINFOMA INFECCIONES |
| url | https://hdl.handle.net/20.500.12008/52138 |