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1.
001-es BibID:
BIBFORM115775
035-os BibID:
(cikkazonosító)102062 (scopus)85164311201 (wos)001040655800001
Első szerző:
Brito-Zerón, Pilar
Cím:
Mortality risk factors in primary Sjögren syndrome : a real-world, retrospective, cohort study / Brito-Zerón Pilar, Flores-Chávez Alejandra, Horváth Ildiko Fanny, Rasmussen Astrid, Li Xiaomei, Olsson Peter, Vissink Arjan, Priori Roberta, Armagan Berkan, Hernandez-Molina Gabriela, Praprotnik Sonja, Quartuccio Luca, Inanc Nevsun, Özkiziltas Burcugül, Bartoloni Elena, Sebastian Agata, Romao Vasco C., Solans Roser, Pasoto Sandra G., Rischmueller Maureen, Galisteo Carlos, Suzuki Yasunori, Trevisani Virginia Fernandes Moca, Fugmann Cecilia, González-García Andrés, Carubbi Francesco, Jurcut Ciprian, Shimizu Toshimasa, Retamozo Soledad, Atzeni Fabiola, Hofauer Benedikt, Melchor-Díaz Sheila, Gheita Tamer, López-Dupla Miguel, Fonseca-Aizpuru Eva, Giacomelli Roberto, Vázquez Marcos, Consani Sandra, Akasbi Miriam, Nakamura Hideki, Szántó Antónia, Farris A. Darise, Wang Li, Mandl Thomas, Gattamelata Angelica, Kilic Levent, Pirkmajer Katja Perdan, Abacar Kerem, Tufan Abdurrahman, de Vita Salvatore, Bootsma Hendrika, Ramos-Casals Manuel, Sjögren Big Data Consortium
Dátum:
2023
ISSN:
2589-5370
Megjegyzések:
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.
Tárgyszavak:
Orvostudományok
Klinikai orvostudományok
idegen nyelvű folyóiratközlemény külföldi lapban
folyóiratcikk
Cardiovascular
Infection
Lymphoma
Mortality
Sjögren syndrome
Systemic disease
Megjelenés:
eClinicalMedicine. - 61 (2023), p. 1-16. -
További szerzők:
Flores-Chávez, Alejandra
Horváth Ildikó Fanny (1980-) (belgyógyász, allergológus, klinikai immunológus)
Rasmussen, Astrid
Li, Xiaomei
Olsson, Peter
Vissink, Arjan
Priori, Roberta
Armagan, Berkan
Hernandez-Molina, Gabriela
Praprotnik, Sonja
Quartuccio, Luca
Inanc, Nevsun
Özkiziltaș, Burcugül
Bartoloni, Elena
Sebastian, Agata
Romão, Vasco C.
Solans, Roser
Pasoto, Sandra
Rischmueller, Maureen
Galisteo, Carlos
Suzuki, Yasunori
Trevisani, Virginia Fernandes Moça
Fugmann, Cecilia
González-García, Andrés
Carubbi, Francesco
Jurcut, Ciprian
Shimizu, Toshimasa
Retamozo, Soledad
Atzeni, Fabiola
Hofauer, Benedikt
Melchor Díaz, S.
Gheita, Tamer A.
López-Dupla, Miguel
Fonseca-Aizpuru, Eva
Giacomelli, Roberto
Vázquez, Marcos
Consani, Sandra
Akasbi, Miriam
Nakamura, Hideki
Szántó Antónia (1977-) (belgyógyász, allergológus és klinikai immunológus)
Farris, Darise
Wang, Li
Mandl, Thomas
Gattamelata, Angelica
Kilic, Levent
Pirkmajer, Katja Perdan
Abacar, Kerem
Tufan, Abdurrahman
Vita, Salvatore de
Bootsma, Hendrika
Ramos-Casals, Manuel
Sjögren Big Data Consortium
Internet cím:
Szerző által megadott URL
DOI
Intézményi repozitóriumban (DEA) tárolt változat
Borító:
Saját polcon:
2.
001-es BibID:
BIBFORM083560
035-os BibID:
(WOS)000574394600030 (Scopus)85078897832
Első szerző:
Brito-Zerón, Pilar
Cím:
Epidemiological profile and north-south gradient driving baseline systemic involvement of primary Sjögren's syndrome / Pilar Brito-Zerón, Nihan Acar-Denizli, Wan-Fai Ng, Ildiko Fanny Horváth, Astrid Rasmussen, Raphaele Seror, Xiaomei Li, Chiara Baldini, Jacques-Eric Gottenberg, Debashish Danda, Luca Quartuccio, Roberta Priori, Gabriela Hernandez-Molina, Berkan Armagan, Aike A. Kruize, Seung-Ki Kwok, Marika Kvarnstrom, Sonja Praprotnik, Damien Sene, Roberto Gerli, Roser Solans, Maureen Rischmueller, Thomas Mandl, Yasunori Suzuki, David Isenberg, Valeria Valim, Piotr Wiland, Gunnel Nordmark, Guadalupe Fraile, Hendrika Bootsma, Hideki Nakamura, Roberto Giacomelli, Valerie Devauchelle-Pensec, Benedikt Hofauer, Michele Bombardieri, Virginia Fernandes Moça Trevisani, Daniel Hammenfors, Sandra G. Pasoto, Soledad Retamozo, Tamer A. Gheita, Fabiola Atzeni, Jacques Morel, Cristina Vollenweider, Margit Zeher, Kathy Sivils, Bei Xu, Stefano Bombardieri, Pulukool Sandhya, Salvatore De Vita, Antonina Minniti, Jorge Sánchez-Guerrero, Levent Kilic, Eefje van der Heijden, Sung-Hwan Park, Marie Wahren-Herlenius, Xavier Mariette, Manuel Ramos-Casals, Sjögren Big Data Consortium
Dátum:
2020
ISSN:
1462-0324 1462-0332
Megjegyzések:
OBJECTIVE: To characterize the systemic phenotype of primary Sjögren's syndrome at diagnosis by analysing the EULAR-SS disease activity index (ESSDAI) scores. METHODS: The Sjögren Big Data Consortium is an international, multicentre registry based on worldwide data-sharing cooperative merging of pre-existing databases from leading centres in clinical research in Sjögren's syndrome from the five continents. RESULTS: The cohort included 10 007 patients (9352 female, mean 53?years) with recorded ESSDAI scores available. At diagnosis, the mean total ESSDAI score was 6.1; 81.8% of patients had systemic activity (ESSDAI score ?1). Males had a higher mean ESSDAI (8.1 vs 6.0, P?<?0.001) compared with females, as did patients diagnosed at <35?years (6.7 vs 5.6 in patients diagnosed at >65?years, P?<?0.001). The highest global ESSDAI score was reported in Black/African Americans, followed by White, Asian and Hispanic patients (6.7, 6.5, 5.4 and 4.8, respectively; P?<?0.001). The frequency of involvement of each systemic organ also differed between ethnic groups, with Black/African American patients showing the highest frequencies in the lymphadenopathy, articular, peripheral nervous system, CNS and biological domains, White patients in the glandular, cutaneous and muscular domains, Asian patients in the pulmonary, renal and haematological domains and Hispanic patients in the constitutional domain. Systemic activity measured by the ESSDAI, clinical ESSDAI (clinESSDAI) and disease activity states was higher in patients from southern countries (P?<?0.001). CONCLUSION: The systemic phenotype of primary Sjögren's syndrome is strongly influenced by personal determinants such as age, gender, ethnicity and place of residence, which are key geoepidemiological players in driving the expression of systemic disease at diagnosis.
Tárgyszavak:
Orvostudományok
Klinikai orvostudományok
idegen nyelvű folyóiratközlemény külföldi lapban
folyóiratcikk
ethnicity
gender
geoepidemiology
phenotype
primary Sjögren's syndrome
Megjelenés:
Rheumatology. - 59 : 9 (2020), p. 2350-2359. -
További szerzők:
Acar-Denizli, Nihan
Ng, Wan Fai
Horváth Ildikó Fanny (1980-) (belgyógyász, allergológus, klinikai immunológus)
Rasmussen, Astrid
Seror, Raphaele
Li, Xiaomei
Baldini, Chiara
Gottenberg, Jacques-Eric
Danda, Debashish
Quartuccio, Luca
Priori, Roberta
Hernandez-Molina, Gabriela
Armagan, Berkan
Kruize, Aike A.
Kwok, Seung-Ki
Kvarnstrom, Marika
Praprotnik, Sonja
Sene, Damien
Gerli, Roberto
Solans, Roser
Rischmueller, Maureen
Mandl, Thomas
Suzuki, Yasunori
Isenberg, David A.
Valim, Valeria
Wiland, Piotr
Nordmark, Gunnel
Fraile, Guadalupe
Bootsma, Hendrika
Nakamura, Hideki
Giacomelli, Roberto
Devauchelle-Pensec, Valerie
Hofauer, Benedikt
Bombardieri, Michele
Trevisani, Virginia Fernandes Moça
Hammenfors, Daniel
Pasoto, Sandra
Retamozo, Soledad
Gheita, Tamer A.
Atzeni, Fabiola
Morel, Jacques
Vollenveider, Cristina
Zeher Margit (1957-2018) (belgyógyász, allergológus és klinikai immunológus, reumatológus)
Sivils, Kathy
Xu, Bei
Bombardieri, Stefano
Sandhya, Pulukool
Vita, Salvatore de
Minniti, Antonina
Sanchez-Guerrero, Jorge
Kilic, Levent
Heijden, Eefje van der
Park, Sung-Hwan
Wahren-Herlenius, Marie
Mariette, Xavier
Ramos-Casals, Manuel
Sjögren Big Data Consortium
Internet cím:
Szerző által megadott URL
DOI
Intézményi repozitóriumban (DEA) tárolt változat
Borító:
Saját polcon:
3.
001-es BibID:
BIBFORM074743
035-os BibID:
(WOS)000446486100015 (Scopus)85055613559
Első szerző:
Brito-Zerón, Pilar
Cím:
How immunological profile drives clinical phenotype of primary Sjögren's syndrome at diagnosis : analysis of 10,500 patients (Sjögren Big Data Project) / P. Brito-Zerón, N. Acar-Denizli, W. F. Ng, M. Zeher, A. Rasmussen, T. Mandl, R. Seror, X. Li, C. Baldini, Jacques-Eric Gottenberg, D. Danda, L. Quartuccio, R. Priori, G. Hernandez-Molina, B. Armagan, A. A. Kruize, Seung-Ki Kwok, M. Kvarnström, S. Praprotnik, D. Sène, E. Bartoloni, R. Solans, M. Rischmueller, Y. Suzuki, D. A. Isenberg, V. Valim, P. Wiland, G. Nordmark, G. Fraile, H. Bootsma, T. Nakamura, R. Giacomelli, V. Devauchelle-Pensec, A. Knopf, M. Bombardieri, V. Trevisani, D. Hammenfors, S. Pasoto, S. Retamozo, T. A. Gheita, F. Atzeni, J. Morel, C. Vollenveider, I. Horvath, K. Sivils, P. Olsson, S. De Vita, J. Sánchez-Guerrero, L. Kilic, M. Wahren-Herlenius, X. Mariette, M. Ramos-Casals, Sjögren Big Data Consortium
Dátum:
2018
ISSN:
0392-856X
Megjegyzések:
OBJECTIVES:To evaluate the influence of the main immunological markers on the disease phenotype at diagnosis in a large international cohort of patients with primary Sjögren's syndrome (SjS). METHODS:The Big Data Sjögren Project Consortium is an international, multicentre registry created in 2014. As a first step, baseline clinical information from leading centres on clinical research in SjS of the 5 continents was collected. The centres shared a harmonised data architecture and conducted cooperative online efforts in order to refine collected data under the coordination of a big data statistical team. Inclusion criteria were the fulfillment of the 2002 classification criteria. Immunological tests were carried out using standard commercial assays. RESULTS:By January 2018, the participant centres had included 10,500 valid patients from 22 countries. The cohort included 9,806 (93%) women and 694 (7%) men, with a mean age at diagnosis of primary SjS of 53 years, mainly White (78%) and included from European countries (71%). The frequency of positive immunological markers at diagnosis was 79.3% for ANA, 73.2% for anti-Ro, 48.6% for RF, 45.1% for anti- La, 13.4% for low C3 levels, 14.5% for low C4 levels and 7.3% for cryoglobulins. Positive autoantibodies (ANA, Ro, La) correlated with a positive result in salivary gland biopsy, while hypocomplementaemia and especially cryoglo-bulinaemia correlated with systemic activity (mean ESSDAI score of 17.7 for cryoglobulins, 11.3 for low C3 and 9.2 for low C4, in comparison with 3.8 for negative markers). The immunological markers with a great number of statistically-significant associations (p<0.001) in the organ-by-organ ESS- DAI evaluation were cryoglobulins (9 domains), low C3 (8 domains), anti-La (7 domains) and low C4 (6 domains). CONCLUSIONS:We confirm the strong influence of immunological markers on the phenotype of primary SjS at diagnosis in the largest multi-ethnic international cohort ever analysed, with a greater influence for cryoglobulinaemic-related markers in comparison with Ro/La autoantibodies and ANA. Immunological patterns play a central role in the phenotypic expression of the disease already at the time of diagnosis, and may guide physicians to design a specific personalised management during the follow-up of patients with primary SjS.
Tárgyszavak:
Orvostudományok
Klinikai orvostudományok
idegen nyelvű folyóiratközlemény külföldi lapban
folyóiratcikk
primary Sjögren's syndrome
salivary gland biopsy
Ro/La autoantibodies
hypocomplementaemia
cryoglobulinaemia
ESSDAI
Megjelenés:
Clinical and Experimental Rheumatology. - 36 : Suppl. 112 (2018), p. S102-S112. -
További szerzők:
Acar-Denizli, Nihan
Ng, Wan Fai
Zeher Margit (1957-2018) (belgyógyász, allergológus és klinikai immunológus, reumatológus)
Rasmussen, Astrid
Mandl, Thomas
Seror, Raphaele
Li, X.
Baldini, Chiara
Gottenberg, Jacques-Eric
Danda, Debashish
Quartuccio, Luca
Priori, Roberta
Hernandez-Molina, Gabriela
Armagan, Berkan
Kruize, Aike A.
Kwok, Seung-Ki
Kvarnstrom, Marika
Praprotnik, Sonja
Sene, Damien
Bartoloni, Elena
Solans, Roser
Rischmueller, Maureen
Suzuki, Yasunori
Isenberg, David A.
Valim, Valeria
Wiland, Piotr
Nordmark, Gunnel
Fraile, Guadalupe
Bootsma, Hendrika
Nakamura, T.
Giacomelli, Roberto
Devauchelle-Pensec, Valerie
Knopf, A.
Bombardieri, Michele
Trevisani, Virginia Fernandes Moça
Hammenfors, Daniel
Pasoto, Sandra
Retamozo, Soledad
Gheita, Tamer A.
Atzeni, F.
Morel, Jacques
Vollenveider, Cristina
Horváth Ildikó Fanny (1980-) (belgyógyász, allergológus, klinikai immunológus)
Sivils, Kathy
Olsson, Peter
Vita, Salvatore de
Sanchez-Guerrero, Jorge
Kilic, Levent
Wahren-Herlenius, Marie
Mariette, Xavier
Ramos-Casals, Manuel
Sjögren Big Data Consortium
Internet cím:
Szerző által megadott URL
Intézményi repozitóriumban (DEA) tárolt változat
Borító:
Saját polcon:
4.
001-es BibID:
BIBFORM100739
035-os BibID:
(WOS)000753131900023 (Scopus)85113866014
Első szerző:
Ramos-Casals, Manuel
Cím:
Childhood-onset of primary Sjögren's syndrome : phenotypic characterization at diagnosis of 158 children / Ramos-Casals Manuel, Acar-Denizli Nihan, Vissink Arjan, Brito-Zerón Pilar, Li Xiaomei, Carubbi Francesco, Priori Roberta, Toplak Nataŝa, Baldini Chiara, Faugier-Fuentes Enrique, Kruize Aike A., Mandl Thomas, Tomiita Minako, Gandolfo Saviana, Hashimoto Kunio, Hernandez-Molina Gabriela, Hofauer Benedikt, Mendieta-Zerón Samara, Rasmussen Astrid, Sandhya Pulukool, Sene Damien, Trevisani Virginia Fernandes Moca, Isenberg David, Sundberg Erik, Pasoto Sandra G., Sebastian Agata, Suzuki Yasunori, Retamozo Soledad, Xu Bei, Giacomelli Roberto, Gattamelata Angelica, Bizjak Masa, Bombardieri Stefano, Loor-Chavez Richard-Eduardo, Hinrichs Anneline, Olsson Peter, Bootsma Hendrika, Lieberman Scott M., Sjogren Big Data Consortium
Dátum:
2021
ISSN:
1462-0324 1462-0332
Megjegyzések:
Objectives: To characterize the phenotypic presentation at diagnosis of childhood-onset primary SS. Methods: The Big Data Sjögren Project Consortium is an international, multicentre registry using worldwide data-sharing cooperative merging of pre-existing clinical SS databases from the five continents. For this study, we selected those patients in whom the disease was diagnosed below the age of 19 years according to the fulfilment of the 2002/2016 classification criteria. Results: Among the 12 083 patients included in the Sjögren Big Data Registry, 158 (1.3%) patients had a childhood-onset diagnosis (136 girls, mean age of 14.2 years): 126 (80%) reported dry mouth, 111 (70%) dry eyes, 52 (33%) parotid enlargement, 118/122 (97%) positive minor salivary gland biopsy and 60/64 (94%) abnormal salivary US study, 140/155 (90%) positive ANA, 138/156 (89%) anti-Ro/La antibodies and 86/142 (68%) positive RF. The systemic EULAR Sjögren's syndrome disease activity index (ESSDAI) domains containing the highest frequencies of active patients included the glandular (47%), articular (26%) and lymphadenopathy (25%) domains. Patients with childhood-onset primary SS showed the highest mean ESSDAI score and the highest frequencies of systemic disease in 5 (constitutional, lymphadenopathy, glandular, cutaneous and haematological) of the 12 ESSDAI domains, and the lowest frequencies in 4 (articular, pulmonary, peripheral nerve and CNS) in comparison with patients with adult-onset disease. Conclusions: Childhood-onset primary SS involves around 1% of patients with primary SS, with a clinical phenotype dominated by sicca features, parotid enlargement and systemic disease. Age at diagnosis plays a key role in modulating the phenotypic expression of the disease.
Tárgyszavak:
Orvostudományok
Klinikai orvostudományok
idegen nyelvű folyóiratközlemény külföldi lapban
folyóiratcikk
Sjogren's syndrome
autoimmune diseases
childhood
epidemiology
paediatrics
Megjelenés:
Rheumatology. - 60 : 10 (2021), p. 4558-4567. -
További szerzők:
Acar-Denizli, Nihan
Vissink, Arjan
Brito-Zerón, Pilar
Li, Xiaomei
Carubbi, Francesco
Priori, Roberta
Toplak, Nataŝa
Baldini, Chiara
Faugier-Fuentes, Enrique
Kruize, Aike A.
Mandl, Thomas
Tomiita, Minako
Gandolfo, Saviana
Hashimoto, Kunio
Hernandez-Molina, Gabriela
Hofauer, Benedikt
Mendieta-Zerón, Samara
Rasmussen, Astrid
Sandhya, Pulukool
Sene, Damien
Trevisani, Virginia Fernandes Moça
Isenberg, David A.
Sundberg, Erik
Pasoto, Sandra
Sebastian, Agata
Suzuki, Yasunori
Retamozo, Soledad
Xu, Bei
Giacomelli, Roberto
Gattamelata, Angelica
Bizjak, Masa
Bombardieri, Stefano
Loor-Chavez, Richard-Eduardo
Hinrichs, Anneline
Olsson, Peter
Bootsma, Hendrika
Lieberman, Scott M.
Horváth Ildikó Fanny (1980-) (belgyógyász, allergológus, klinikai immunológus)
Szántó Antónia (1977-) (belgyógyász, allergológus és klinikai immunológus)
Sjögren Big Data Consortium
Internet cím:
Szerző által megadott URL
DOI
Intézményi repozitóriumban (DEA) tárolt változat
Borító:
Saját polcon:
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