Occurrence of progressive pulmonary fibrosis and its association with survival in connective tissue disease-associated interstitial lung disease: an international retrospective cohort study. - 2026
Occurrence of progressive pulmonary fibrosis and its association with survival in connective tissue disease-associated interstitial lung disease: an international retrospective cohort study.
Guiot, Julien; Teitsma, Xavier M; Dimitroulas, Theodoroset al.
[en] To describe real-world management of connective tissue disease-associated interstitial lung disease (CTD-ILD) and explore associations between progressive pulmonary fibrosis (PPF) onset and clinical outcomes. This international, retrospective cohort included adults with rheumatoid arthritis (RA), systemic sclerosis (SSc), mixed connective tissue disease, and other CTD with associated ILD. PPF was identified using INBUILD and/or ATS/ERS/JRS/ALAT criteria applied in routine care. Analyses were descriptive and exploratory; time-to-event methods assessed selected outcomes and factors associated with PPF onset. Among 357 patients (mean age 61.6 years; 58% female), SSc-ILD (31%) and RA-ILD (29%) were most common. At ILD diagnosis, 21.6% had no respiratory symptoms. During a mean follow-up of 60.5 months, 147 patients (41.2%) met the PPF criteria, at a median of 30.6 months after CTD-ILD diagnosis. Following PPF onset, the most frequently prescribed therapies were corticosteroids (72.8%), mycophenolate mofetil (52.4%), and antifibrotics (51.0%). Lung function data were available for 37.8% at diagnosis and 53.7% at year 5. PPF was associated with greater observed lung function decline compared to non-PPF patients. ILD-related hospitalizations was numerically more frequent within PPF patients (37.4% vs. 28.6%), but the difference was not statistically significant (P = 0.078). Among patients with PPF, earlier versus later onset, defined by a subtype-specific median split, was associated with poorer observed survival (P < 0.001). In this retrospective cohort, PPF was observed frequently in CTD-ILD patients, particularly those with SSc-ILD, and was associated with greater lung function decline and higher healthcare utilisation. Earlier PPF onset was associated with higher mortality risk, but missing lung function data, surveillance differences, time-related bias, and residual confounding limit causal interpretation. Prospective studies using harmonised PPF adjudication and time-dependent analyses are needed.
Disciplines :
Cardiovascular & respiratory systems
Author, co-author :
Guiot, Julien ; Université de Liège - ULiège > Département des sciences cliniques > Pneumologie - Allergologie
Teitsma, Xavier M ; Boehringer Ingelheim B.V., Basisweg 10, 1043 AP, Amsterdam, The Netherlands. xavier.teitsma@boehringer-ingelheim.com
Dimitroulas, Theodoros ; 4th Department of Internal Medicine, Hippokration Hospital, School of Medicine, Aristotle University of Thessaloniki, Thessaloniki, Greece
Davidsen, Jesper Rømhild ; South Danish Center for Interstitial Lung Diseases (SCILS), Department of Respiratory Medicine, Odense University Hospital, Odense, Denmark
Patabendige, Sanjeewa ; South Danish Center for Interstitial Lung Diseases (SCILS), Department of Respiratory Medicine, Odense University Hospital, Odense, Denmark
Duarte, Ana Catarina ; Department of Rheumatology, Hospital Garcia de Orta, Unidade Local de Saúde de Almada-Seixal, Almada, Portugal
Prata, Ana Rita Luis Alves ; Department of Rheumatology, Unidade Local de Saúde Região de Aveiro, Ovar, Portugal
Wuyts, Wim A ; Unit for Interstitial Lung Diseases, Department of Respiratory Medicine, University Hospitals Leuven, Leuven, Belgium
Language :
English
Title :
Occurrence of progressive pulmonary fibrosis and its association with survival in connective tissue disease-associated interstitial lung disease: an international retrospective cohort study.
Althobiani MA Russell AM Jacob J Ranjan Y Folarin AA Hurst JR et al. Interstitial lung disease: a review of classification, etiology, epidemiology, clinical diagnosis, pharmacological and non-pharmacological treatment Front Med (Lausanne) 2024 11 1296890 10.3389/fmed.2024.1296890 38698783 11063378
Hyldgaard C Bendstrup E Pedersen AB Pedersen L Ellingsen T Interstitial Lung Disease in Connective Tissue Diseases: Survival Patterns in a Population-Based Cohort J Clin Med 2021 10 21 4830 10.3390/jcm10214830 34768349 8584507
Panagopoulos P Goules A Hoffmann-Vold AM Matteson EL Tzioufas A Natural history and screening of interstitial lung disease in systemic autoimmune rheumatic disorders Ther Adv Musculoskelet Dis 2021 13 1759720X211037519 1:CAS:528:DC%2BB38Xhs1Srtg%3D%3D 10.1177/1759720X211037519 34471427 8404673
Zhuo J Bao Y Xia Q Rao A Samal C Lama S Interstitial Lung Disease Associated Health Care Resource Utilization and Cost in Rheumatoid Arthritis Patients in an Insured Population Arthritis Rheumatol 2019 71 Suppl 10 Abstract254
Shao T Shi X Yang S Zhang W Li X Shu J et al. Interstitial Lung Disease in Connective Tissue Disease: A Common Lesion With Heterogeneous Mechanisms and Treatment Considerations Front Immunol 2021 12 684699 1:CAS:528:DC%2BB3MXhs1SqtbvN 10.3389/fimmu.2021.684699 34163483 8215654
Cosgrove GP Bianchi P Danese S Lederer DJ Barriers to timely diagnosis of interstitial lung disease in the real world: the INTENSITY survey BMC Pulm Med 2018 18 1 9 10.1186/s12890-017-0560-x 29343236 5773175
Johnson SR Bernstein EJ Bolster MB Chung JH Danoff SK George MD et al. 2023 American College of Rheumatology (ACR)/American College of Chest Physicians (CHEST) Guideline for the Treatment of Interstitial Lung Disease in People with Systemic Autoimmune Rheumatic Diseases Arthritis Care Res (Hoboken) 2024 76 8 1051 1069 10.1002/acr.25348 38973731 12646475
Antoniou KM Distler O Gheorghiu AM Moor CC Vikse J Bizymi N et al. ERS/EULAR clinical practice guidelines for connective tissue diseases associated interstitial lung disease Ann Rheum Dis 2025 S0003–4967 25 04320 04321
Johnson SR Bernstein EJ Bolster MB Chung JH Danoff SK George MD et al. 2023 American College of Rheumatology (ACR)/American College of Chest Physicians (CHEST) Guideline for the Screening and Monitoring of Interstitial Lung Disease in People with Systemic Autoimmune Rheumatic Diseases Arthritis Care Res 2024 76 8 1070 1082 10.1002/acr.25347
Antoniou KM, Distler O, Gheorghiu AM, Moor CC, Vikse J, Bizymi N et al (2025) ERS/EULAR clinical practice guidelines for connective tissue disease-associated interstitial lung disease. Developed by the task force for connective tissue disease-associated interstitial lung disease of the European Respiratory Society (ERS) and the European Alliance of Associations for Rheumatology (EULAR) Endorsed by the European Reference Network on rare respiratory diseases (ERN-LUNG). Eur Respir J 2500896
Hoffmann-Vold AM Maher TM Philpot EE Ashrafzadeh A Barake R Barsotti S et al. The identification and management of interstitial lung disease in systemic sclerosis: evidence-based European consensus statements Lancet Rheumatol 2020 2 2 e71 83 1:CAS:528:DC%2BB2MXot1Sntbw%3D 10.1016/S2665-9913(19)30144-4 38263663
Yang J Head MA Schott KD Tan H Djaraher R Crowe CL et al. Real-world care patterns and specialist encounters of patients with systemic autoimmune rheumatic disease-related interstitial lung disease in the United States: a retrospective administrative claims database analysis Rheumatology (Oxford) 2025 64 8 4713 4721 10.1093/rheumatology/keaf200 40268746 12316361
Hannah J Rodziewicz M Mehta P Heenan KM Ball E Barratt S et al. The diagnosis and management of systemic autoimmune rheumatic disease-related interstitial lung disease: British Society for Rheumatology guideline scope Rheumatol Adv Pract 2024 8 2 rkae056 10.1093/rap/rkae056 38765189 11101284
Doyle TJ Hunninghake GM Rosas IO Subclinical Interstitial Lung Disease Am J Respir Crit Care Med 2012 185 11 1147 1153 10.1164/rccm.201108-1420PP 22366047 3373068
Drimus JC Duma RC Trăilă D Mogoșan CD Manolescu DL Fira-Mladinescu O High-Resolution CT Findings in Interstitial Lung Disease Associated with Connective Tissue Diseases: Differentiating Patterns for Clinical Practice-A Systematic Review with Meta-Analysis J Clin Med 2025 14 17 6164 1:CAS:528:DC%2BB2MXitlSmsb3L 10.3390/jcm14176164 40943925 12429569
Vij R Strek ME Diagnosis and Treatment of Connective Tissue Disease-Associated Interstitial Lung Disease Chest 2013 143 3 814 824 1:CAS:528:DC%2BC3sXltVaqs7o%3D 10.1378/chest.12-0741 23460159 3590889
Rajan SK Cottin V Dhar R Danoff S Flaherty KR Brown KK et al. Progressive pulmonary fibrosis: an expert group consensus statement Eur Respir J 2023 61 3 2103187 10.1183/13993003.03187-2021 36517177 10060665
Wijsenbeek M Kreuter M Olson A Fischer A Bendstrup E Wells CD et al. Progressive fibrosing interstitial lung diseases: current practice in diagnosis and management Curr Med Res Opin 2019 35 11 2015 2024 10.1080/03007995.2019.1647040 31328965
Spagnolo P Ryerson CJ Putman R Oldham J Salisbury M Sverzellati N et al. Early diagnosis of fibrotic interstitial lung disease: challenges and opportunities Lancet Respiratory Med 2021 9 9 1065 1076 1:CAS:528:DC%2BB3MXisFSjs7bJ 10.1016/S2213-2600(21)00017-5
Fabbri L Russell AM Chaudhuri N Adams W Cowan K Conway J et al. Research priorities for progressive pulmonary fibrosis in the UK BMJ Open Respir Res 2024 11 1 e002368 10.1136/bmjresp-2024-002368 39231598 11428979
Liakouli V Ciancio A Del Galdo F Giacomelli R Ciccia F Systemic sclerosis interstitial lung disease: unmet needs and potential solutions Nat Rev Rheumatol 2024 20 1 21 32 10.1038/s41584-023-01044-x 37923862
Flaherty KR Wells AU Cottin V Devaraj A Walsh SLF Inoue Y et al. Nintedanib in Progressive Fibrosing Interstitial Lung Diseases N Engl J Med 2019 381 18 1718 1727 1:CAS:528:DC%2BC1MXitFWktrzL 10.1056/NEJMoa1908681 31566307
Raghu G Remy-Jardin M Richeldi L Thomson CC Inoue Y Johkoh T et al. Idiopathic Pulmonary Fibrosis (an Update) and Progressive Pulmonary Fibrosis in Adults: An Official ATS/ERS/JRS/ALAT Clinical Practice Guideline Am J Respir Crit Care Med 2022 205 9 e18 47 10.1164/rccm.202202-0399ST 35486072 9851481
Wells AU Walsh SLF Adegunsoye A Cottin V Danoff SK Devaraj A et al. Identification of progressive pulmonary fibrosis: consensus findings from a modified Delphi study Respir Res 2024 25 1 448 1:CAS:528:DC%2BB2MXlsVGitQ%3D%3D 10.1186/s12931-024-03070-z 39741294 11687192
Podolanczuk AJ Hunninghake GM Wilson KC Khor YH Kheir F Pang B et al. Approach to the Evaluation and Management of Interstitial Lung Abnormalities: An Official American Thoracic Society Clinical Statement Am J Respir Crit Care Med 2025 211 7 1132 1155 1:CAS:528:DC%2BB2MXit1aks7nI 10.1164/rccm.202505-1054ST 40387336 12264694
Wagih Abdelwahab H Shalabi NM Mohamed Rashad Ghoneim M Saad Farrag N Hamdy F Elhoseiny F et al. Screening for Subclinical Interstitial Lung Disease in Rheumatoid Arthritis Patients: Functional and Radiological Methods Turk Thorac J 2022 23 4 261 267 10.5152/TurkThoracJ.2022.21263 9361088
Hong M Veitch E Limaye S Connective tissue disease-associated interstitial lung disease: an approach to treatment amidst an expanding evidence base Intern Med J 2025 10.1111/imj.70026 41451789 12155064
Chang SH Lee JS Ha YJ Kim MU Park CH Lee JS et al. Lung function trajectory of rheumatoid arthritis–associated interstitial lung disease Rheumatology (Oxford) 2023 62 9 3014 3024 1:CAS:528:DC%2BB2cXhsFemu7w%3D 10.1093/rheumatology/kead027 36702465 10473227
Chan C Ryerson CJ Dunne JV Wilcox PG Demographic and clinical predictors of progression and mortality in connective tissue disease-associated interstitial lung disease: a retrospective cohort study BMC Pulm Med 2019 19 1 192 10.1186/s12890-019-0943-2 31672127 6824100
Molina-Molina M Castellví I Valenzuela C Ramirez J Rodríguez Portal JA Franquet T et al. Management of progressive pulmonary fibrosis associated with connective tissue disease Expert Rev Respir Med 2022 16 7 765 774 1:CAS:528:DC%2BB38XitVKgtbbP 10.1080/17476348.2022.2107508 35912842
Montero IE Hernandez-Gonzalez F Sellares J Epidemiology and Prognosis of Progressive Pulmonary Fibrosis: A Literature Review Pulm Ther 2025 11 3 347 363 10.1007/s41030-025-00302-5 40522429 12373627
Kondoh Y Ito T Saito K Bao H Sakamoto W Progressive pulmonary fibrosis (PPF): Estimation of incidence and treatment rates in Japan using a claims database Respiratory Invest 2024 62 4 702 709 1:CAS:528:DC%2BB2cXht1GlsL3F 10.1016/j.resinv.2024.05.005
Maher TM Assassi S Azuma A Cottin V Hoffmann-Vold AM Kreuter M et al. Nerandomilast in Patients with Progressive Pulmonary Fibrosis N Engl J Med 2025 392 22 2203 2214 1:CAS:528:DC%2BB2MXhsVCmsrfP 10.1056/NEJMoa2503643 40388329
Nasser M Larrieu S Boussel L Si-Mohamed S Bazin F Marque S et al. Estimates of epidemiology, mortality and disease burden associated with progressive fibrosing interstitial lung disease in France (the PROGRESS study) Respir Res 2021 22 1 162 10.1186/s12931-021-01749-1 34030695 8147348
Behr J Kreuter M Guenther AU Bonella F Pittrow D Pausch C et al. Real-life characteristics and management of patients with fibrosing interstitial lung disease: INSIGHTS-ILD registry ERJ Open Res 2025 11 3 00926 2024 10.1183/23120541.00926-2024 40491463 12147108
Pires FS Damas C Mota P Melo N Costa D Jesus JM et al. Slow versus rapid progressors in idiopathic pulmonary fibrosis Eur Respir J 2011 38 Suppl 55 P656 10.1183/13993003/erj.38.Suppl_55.p656