ArticleRheumatology (Oxford, England)2023
Anti-FHL1 autoantibodies in juvenile myositis are associated with anti-Ro52 autoantibodies but not with severe disease features.
Matthew A Sherman, Rose Graf, Sara E Sabbagh, Angeles S Galindo-Feria, Iago Pinal-Fernandez, Katherine Pak, Takayuki Kishi, Willy A Flegel, Ira N Targoff, Frederick W Miller and 4 more
Open access · hybridAbstract read
In one paragraphArticle in Rheumatology (Oxford, England), 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
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What it found
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3 · Its place in the literatureWho cites it
0 citing papers in PubMed, 4 citations in OpenAlex.
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4 · The recordCorrections and comments
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5 · Who and what moneyAuthors and funding
14 authors at 6 institutions in 2 countries.
Matthew A ShermanMuscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD.ORCID 0000-0002-5448-1538 Rose GrafMuscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD.
Sara E SabbaghDivision of Rheumatology, Department of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA.
Angeles S Galindo-FeriaDivision of Rheumatology, Department of Medicine, Solna, Karolinska Institutet.ORCID 0000-0001-7095-211X Iago Pinal-FernandezMuscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD.
Katherine PakMuscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD.
Takayuki KishiEnvironmental Autoimmunity Group, National Institute of Environmental Health Sciences.ORCID 0000-0002-7208-1562 Willy A FlegelDepartment of Transfusion Medicine, NIH Clinical Center, National Institutes of Health, Bethesda, MD.
Ira N TargoffVeteran's Affairs Medical Center, University of Oklahoma Health Sciences Center, and Oklahoma Medical Research Foundation, Oklahoma City, OK.
Frederick W MillerEnvironmental Autoimmunity Group, National Institute of Environmental Health Sciences.
Andrew L MammenMuscle Disease Unit, Laboratory of Muscle Stem Cells and Gene Regulation, National Institute of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health, Bethesda, MD.
Childhood Myositis Heterogeneity Collaborative Study Group
National Institutes of Health · USNational Institute of Environmental Health Sciences · USKarolinska University Hospital · SEMedical College of Wisconsin · USNational Institutes of Health Clinical Center · USOklahoma Medical Research Foundation · US
Funding
Environmental/genetic Risk Factors and Pathogenesis of Autoimmune DiseaseZIAES101074 · NIEHS · NATIONAL INSTITUTE OF ENVIRONMENTAL HEALTH SCIENCES · PI RIDER, LISA · 2009 to 2025
$34.6MMuscle Disease UnitZIAAR041203 · NIAMS · NATIONAL INSTITUTE OF ARTHRITIS AND MUSCULOSKELETAL AND SKIN DISEASES · PI MAMMEN, ANDREW · 2015 to 2025
$19.7MAssessment, Therapy and Prevention Of Autoimmune DiseaseZIAES101081 · NIEHS · NATIONAL INSTITUTE OF ENVIRONMENTAL HEALTH SCIENCES · PI RIDER, LISA · 2009 to 2025
$17.6MLaboratory Services Section, Department of Transfusion Medicine Core ReportZICCL002128 · CLC · CLINICAL CENTER · PI FLEGEL, WILLY · 2020 to 2025
$0kIntramural NIH HHS ZIA AR041203Intramural NIH HHS ZIA ES101074Intramural NIH HHS ZIA ES101081Intramural NIH HHS ZIC CL002128
6 · The paper itselfAbstract
objectivesFour-and-a-half LIM domains 1 (FHL1) is a muscle-specific protein. Autoantibodies against FHL1 were recently discovered in adults with idiopathic inflammatory myopathies (IIMs) and were found to be associated with clinical features and outcomes indicative of increased disease severity. Anti-FHL1 autoantibodies have not been described in children. Here, the prevalence and clinical features associated with anti-FHL1 autoantibodies were examined in a large North American cohort of juvenile patients with IIM.
methodsSera from 338 juvenile IIM patients and 91 juvenile healthy controls were screened for anti-FHL1 autoantibodies by ELISA. Clinical characteristics and HLA alleles of those with and without anti-FHL1 autoantibodies were compared among those with juvenile IIM.
resultsAnti-FHL1 autoantibodies were present in 10.9% of juvenile IIM patients and 1.1% of controls. The frequency of anti-FHL1 autoantibodies among clinical and serologic subgroups did not differ. A higher percentage of Asian patients had anti-FHL1 autoantibodies (11% vs 0.7%; P = 0.002). Myositis-associated autoantibodies (MAAs) [odds ratio (OR) 2.09 (CI 1.03, 4.32)], anti-Ro52 autoantibodies specifically [OR 4.17 (CI 1.83, 9.37)] and V-sign rash [OR 2.59 (CI 1.22, 5.40)] were associated with anti-FHL1 autoantibodies. There were no differences in other features or markers of disease severity. No HLA associations with anti-FHL1 autoantibodies in Caucasian myositis patients were identified.
conclusionAnti-FHL1 autoantibodies are present in ∼11% of juvenile IIM patients and commonly co-occur with MAAs, including anti-Ro52 autoantibodies. In contrast to adult IIM, anti-FHL1 autoantibodies in juvenile myositis are associated with V-sign rash but not with other distinctive clinical features or worse outcomes.
Indexed as
DermatomyositisExanthemaMyositisAdultAntibodies, AntinuclearAutoantibodiesChildHumansIntracellular Signaling Peptides and ProteinsLIM Domain ProteinsMuscle ProteinsAntibodies, AntinuclearAutoantibodiesFHL1 protein, humanIntracellular Signaling Peptides and ProteinsLIM Domain ProteinsMuscle ProteinsSS-A antibodiesFHL1 autoantibodiesjuvenile idiopathic inflammatory myopathiesmyositismyositis-associated autoantibodies
Identifiers
PMID35961028
PMCPMC9949705
OpenAlexW4290930945
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