Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in Taiwan
IntroductionThe clinical characteristics of patients positive for anti-small ubiquitin-like modifier 1-activating enzyme subunit 1 (SAE1) antibodies and diagnosed with idiopathic inflammatory myopathies (IIMs) vary across different cohorts and ethnicities, particularly concerning interstitial lung d...
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Frontiers Media S.A.
2024-11-01
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fimmu.2024.1482000/full |
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| author | Chao-Yang Hsiao Chao-Yang Hsiao Shu-Chi Tseng Chung-Yuan Hsu Li-Chung Chiu Li-Jen Su Tien-Ming Chan |
| author_facet | Chao-Yang Hsiao Chao-Yang Hsiao Shu-Chi Tseng Chung-Yuan Hsu Li-Chung Chiu Li-Jen Su Tien-Ming Chan |
| author_sort | Chao-Yang Hsiao |
| collection | DOAJ |
| description | IntroductionThe clinical characteristics of patients positive for anti-small ubiquitin-like modifier 1-activating enzyme subunit 1 (SAE1) antibodies and diagnosed with idiopathic inflammatory myopathies (IIMs) vary across different cohorts and ethnicities, particularly concerning interstitial lung disease (ILD). We aimed to assess the clinical utility of the line immunoblot assay (LIA) in detecting anti-SAE1 autoantibodies and evaluate the clinical relevance and chronology of ILD development in relation to SAE1 autoantibody positivity among Taiwanese patients.MethodsWe retrospectively conducted a population-based cohort analysis involving 6,496 patients who visited Chang Gung Memorial Health System across Taiwan from May 2018 to December 2021. Patients were assayed for myositis-specific autoantibodies (MSAs) and myositis-associated autoantibodies (MAAs) using the LIA method, and the antinuclear antibody (ANA) indirect immunofluorescence (IIF) method was used to evaluate ANA patterns. Of these, 70 SAE1-positive patients (1.08%) were included and followed up until December 2023. Associations with clinical characteristics and final diagnosis, particularly ILD, were assessed.ResultsAmong the 70 SAE1-positive patients, 10 (14.3%) were strongly positive and 60 (85.7%) were weakly positive. In the strong positive group, 70% (7/10) were diagnosed with IIM, with most (5/7) showing a concordant ANA IIF pattern (speckled type). Six patients presented ILD either before (1/6) or after (5/6) IIM diagnosis; the majority (4/6) were classified as organizing pneumonia. The remaining 30.0% (3/10) had connective tissue disease (CTD) other than IIM without detectable ILD during follow-up, and none demonstrated a concordant ANA IIF pattern. In the weakly positive group, only 5.0% (3/60) had IIM and 3.3% (2/60) had ILD. The positive predictive value for strong positive SAE1 autoantibodies in diagnosing IIM was significantly higher than for weak positives (70.0% vs. 5.0%; p < 0.001).ConclusionsThe study suggests that strong positive SAE1 autoantibodies detected via LIA are more closely associated with IIM compared to weak positive results. A high prevalence of ILD was observed among strong positive Taiwanese patients, indicating the need for prompt screening. Patients with weak positive or discordant ANA IIF results may represent false positives with a lower ILD risk. |
| format | Article |
| id | doaj-art-14b8fc970b4e4f388420bbcb8e3b475e |
| institution | OA Journals |
| issn | 1664-3224 |
| language | English |
| publishDate | 2024-11-01 |
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| spelling | doaj-art-14b8fc970b4e4f388420bbcb8e3b475e2025-08-20T02:12:48ZengFrontiers Media S.A.Frontiers in Immunology1664-32242024-11-011510.3389/fimmu.2024.14820001482000Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in TaiwanChao-Yang Hsiao0Chao-Yang Hsiao1Shu-Chi Tseng2Chung-Yuan Hsu3Li-Chung Chiu4Li-Jen Su5Tien-Ming Chan6Division of Rheumatology, Allergy and Immunology, Department of Internal Medicine, Chang Gung Memorial Hospital, and Chang Gung University, Taoyuan, TaiwanDepartment of Biomedical Sciences and Engineering, National Central University, Taoyuan, TaiwanDepartment of Medical Imaging and Intervention, Chang Gung Memorial Hospital at Linkou and Chang Gung University, Taoyuan, TaiwanDivision of Rheumatology, Allergy and Immunology, Department of Internal Medicine, Chang Gung Memorial Hospital, Kaohsiung, TaiwanDepartment of Thoracic Medicine, Chang Gung Memorial Hospital, Chang Gung University College of Medicine, Taoyuan, TaiwanDepartment of Biomedical Sciences and Engineering, National Central University, Taoyuan, TaiwanDivision of Rheumatology, Allergy and Immunology, Department of Internal Medicine, Chang Gung Memorial Hospital, and Chang Gung University, Taoyuan, TaiwanIntroductionThe clinical characteristics of patients positive for anti-small ubiquitin-like modifier 1-activating enzyme subunit 1 (SAE1) antibodies and diagnosed with idiopathic inflammatory myopathies (IIMs) vary across different cohorts and ethnicities, particularly concerning interstitial lung disease (ILD). We aimed to assess the clinical utility of the line immunoblot assay (LIA) in detecting anti-SAE1 autoantibodies and evaluate the clinical relevance and chronology of ILD development in relation to SAE1 autoantibody positivity among Taiwanese patients.MethodsWe retrospectively conducted a population-based cohort analysis involving 6,496 patients who visited Chang Gung Memorial Health System across Taiwan from May 2018 to December 2021. Patients were assayed for myositis-specific autoantibodies (MSAs) and myositis-associated autoantibodies (MAAs) using the LIA method, and the antinuclear antibody (ANA) indirect immunofluorescence (IIF) method was used to evaluate ANA patterns. Of these, 70 SAE1-positive patients (1.08%) were included and followed up until December 2023. Associations with clinical characteristics and final diagnosis, particularly ILD, were assessed.ResultsAmong the 70 SAE1-positive patients, 10 (14.3%) were strongly positive and 60 (85.7%) were weakly positive. In the strong positive group, 70% (7/10) were diagnosed with IIM, with most (5/7) showing a concordant ANA IIF pattern (speckled type). Six patients presented ILD either before (1/6) or after (5/6) IIM diagnosis; the majority (4/6) were classified as organizing pneumonia. The remaining 30.0% (3/10) had connective tissue disease (CTD) other than IIM without detectable ILD during follow-up, and none demonstrated a concordant ANA IIF pattern. In the weakly positive group, only 5.0% (3/60) had IIM and 3.3% (2/60) had ILD. The positive predictive value for strong positive SAE1 autoantibodies in diagnosing IIM was significantly higher than for weak positives (70.0% vs. 5.0%; p < 0.001).ConclusionsThe study suggests that strong positive SAE1 autoantibodies detected via LIA are more closely associated with IIM compared to weak positive results. A high prevalence of ILD was observed among strong positive Taiwanese patients, indicating the need for prompt screening. Patients with weak positive or discordant ANA IIF results may represent false positives with a lower ILD risk.https://www.frontiersin.org/articles/10.3389/fimmu.2024.1482000/fullautoantibodyidiopathic inflammatory myopathiesinterstitial lung diseasepositive predictive valueautoimmuneanti-SAE1 |
| spellingShingle | Chao-Yang Hsiao Chao-Yang Hsiao Shu-Chi Tseng Chung-Yuan Hsu Li-Chung Chiu Li-Jen Su Tien-Ming Chan Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in Taiwan Frontiers in Immunology autoantibody idiopathic inflammatory myopathies interstitial lung disease positive predictive value autoimmune anti-SAE1 |
| title | Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in Taiwan |
| title_full | Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in Taiwan |
| title_fullStr | Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in Taiwan |
| title_full_unstemmed | Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in Taiwan |
| title_short | Clinical features of anti-SAE1 antibody-positive myositis and interstitial lung disease: a multicenter, retrospective study in Taiwan |
| title_sort | clinical features of anti sae1 antibody positive myositis and interstitial lung disease a multicenter retrospective study in taiwan |
| topic | autoantibody idiopathic inflammatory myopathies interstitial lung disease positive predictive value autoimmune anti-SAE1 |
| url | https://www.frontiersin.org/articles/10.3389/fimmu.2024.1482000/full |
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