Inborn errors of immunity (IEIs) are immunological disorders characterized by variable susceptibility to infections, immune dysregulation and/or malignancies, as a consequence of damaging germline variants in single genes. Though initially identified among patients with unusual, severe or recurrent infections, non-infectious manifestations and especially immune dysregulation in the form of autoimmunity or autoinflammation can be the first or dominant phenotypic aspect of IEIs. An increasing number of IEIs causing autoimmunity or autoinflammation, including rheumatic disease have been reported over the last decade. Despite their rarity, identification of those disorders provided insight into the pathomechanisms of immune dysregulation, which may be relevant for understanding the pathogenesis of systemic rheumatic disorders. In this review, we present novel IEIs primarily causing autoimmunity or autoinflammation along with their pathogenic mechanisms. In addition, we explore the likely pathophysiological and clinical relevance of IEIs in systemic rheumatic disorders.
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http://dx.doi.org/10.1016/j.ebiom.2023.104501 | DOI Listing |
Eur Arch Otorhinolaryngol
January 2025
Department of Otolaryngology and Head and Neck Surgery, IRCSS AOU San Martino, University of Genoa, Largo Rosanna Benzi 10, 16132, Genoa, Italy.
Purpose: Immunoglobulin G4-related disease (IgG4-RD) is a complex systemic fibroinflammatory condition with different clinical manifestations affecting multiple organ systems. Despite its rarity, the disease presents diagnostic and therapeutic challenges due to its mimicry of malignancies and other immune-mediated disorders. The 2019 American College of Rheumatology/European League Against Rheumatism Classification Criteria for IgG4-Related Disease is the current state of art to confirm the diagnosis of IgG4-RD even in the absence of histological analysis.
View Article and Find Full Text PDFRheumatol Int
January 2025
Pharmacy, Radboudumc, Nijmegen, NL, Netherlands.
This study investigated severity, course and patterns of fatigue surrounding subcutaneous biological disease-modifying antirheumatic drug (bDMARD) injection in inflammatory rheumatic disease (IRD) patients using ecological momentary assessments and investigated self-reported adverse drug reactions (ADRs). In this prospective cohort study, IRD patients completed fatigue severity numeric rating scales (0-10) in web-based ecological momentary assessments in three waves of five days surrounding bDMARD injection. The course of fatigue was measured by the change in fatigue from pre-dosing to post-dosing scores and was classified as: worsening, improving or no clinically relevant change.
View Article and Find Full Text PDFDiabetes Ther
January 2025
First Clinical Medical College, Shandong University of Traditional Chinese Medicine, Department of Endocrinology and Metabology, The First Affiliated Hospital of Shandong First Medical, University & Shandong Provincial Qianfoshan Hospital, Shandong Key Laboratory of Rheumatic Disease and Translational Medicine, Shandong Institute of Nephrology, Jinan, China.
Introduction: More than half of diabetes patients are Asians, and their tolerance to antidiabetic drugs may differ from that of non-Asians. Oral semaglutide has recently gained attention for its advantages in glycemic and body weight control. However, its effects across different ethnic groups remain unknown.
View Article and Find Full Text PDFImmunol Med
January 2025
Department of Diabetes, Endocrinology and Clinical Immunology, Hyogo Medical University School of Medicine, Hyogo, Japan.
Rituximab (RTX) has been reported to effectively maintain remission in anti-neutrophil cytoplasmic antibody-associated vasculitis (AAV). In this multicenter study involving 57 patients who achieved remission after 24 weeks, we evaluated the effectiveness of RTX in maintaining remission in patients with AAV. Patients were divided into three groups based on RTX administration: continuous, induction phase-only, and maintenance phase-only groups.
View Article and Find Full Text PDFInt J Rheum Dis
January 2025
The First Affiliated Hospital of Anhui University of Chinese Medicine, Hefei, Anhui, China.
Background: N6-methyladenosine (m6A) is one of the most conserved internal RNA modifications, which has been implicated in many biological processes, such as apoptosis and proliferation. Wilms tumor 1-associating protein (WTAP), as a key component of m6A methylation, is a nuclear protein that has been associated with the regulation of proliferation and apoptosis. Rheumatoid arthritis (RA), a systemic, infiltrating autoimmune disease, is characterized by synovial hyperplasia.
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