Paroxysmal nocturnal hemoglobinuria (PNH) is an acquired clonal hematologic disorder. Historically, most PNH patients mainly received supportive treatment, which led to poor quality of life and high mortality rates. The advent of downstream complement C5 inhibitors has dramatically changed the natural course of PNH. These inhibitors alleviate clinical symptoms, significantly reduce severe complications, and improve survival rates. Despite their ability to control intravascular hemolysis and enhance hemoglobin levels, approximately half of the treated patients still require blood transfusions due to extravascular hemolysis (EVH) mediated by upstream complement component C3. Upstream complement inhibitors not only control IVH but also reduce the deposition of C3 fragments, thereby solving the problem of EVH and further improving the clinical outcomes of PNH patients. However, due to the cascade of complement activation and the increased accumulation of PNH red blood cells after treatment with upstream complement inhibitors, there may be an increased risk of more severe breakthrough hemolysis events. Additionally, the potential risk of infections associated with complement inhibitors is another issue that needs to be addressed.
Download full-text PDF |
Source |
---|---|
http://dx.doi.org/10.3760/cma.j.cn121090-20240903-00332 | DOI Listing |
Eur J Immunol
March 2025
Department of Immunology, Assistance Publique- Hôpitaux de Paris (AP-HP), Georges Pompidou European Hospital, Paris, France.
Inborn deficiencies of the alternative pathway (AP) of the complement system have been associated with life-threatening infections, mainly by encapsulated bacteria. Complete factor D (FD) deficiencies have been reported in only seven families in the literature. We report two new cases of biochemically and genetically confirmed complete FD deficiency, including the first in a Down syndrome patient.
View Article and Find Full Text PDFRheumatology (Oxford)
March 2025
Department of Medicine, University of Cambridge, Cambridge, UK.
Drug development in ANCA-associated vasculitis has aimed to improve on the success of the B cell depleting monoclonal antibody rituximab and exploit better understanding of inflammatory pathways. More potent B cell depletion strategies are being tested as are B cell cytokine inhibitors. The involvement of the complement system in pathogenesis is more complicated than previously thought and extends beyond C5a dysregulation and its inhibition with avacopan, broader complement inhibitors and complement regulatory agonists are potential newer therapies.
View Article and Find Full Text PDFRheumatology (Oxford)
March 2025
UNC Kidney Center, Division of Nephrology and Hypertension, Department of Medicine, University of North Carolina at Chapel Hill, Chapel Hill, NC, USA.
Our understanding of ANCA vasculitis has advanced from discovery of putative auto-antibodies to a greater understanding of the myriad alterations of innate and adaptive immunity in this disease. The 21st International Vasculitis Workshop held in Barcelona served again as a forum for distributing and sharing advances in this field. B-cell and T-cell subsets are skewed in ANCA vasculitis patients, favoring a pro-inflammatory phenotype.
View Article and Find Full Text PDFRheumatology (Oxford)
March 2025
Vasculitis and Lupus Clinic, Cambridge University Hospitals NHS Foundation Trust, Cambridge, UK.
This session presented emerging realworld evidence on novel glucocorticoid (GC) sparing therapies for ANCA-associated vasculitis (AAV). It covered the first-in-class oral C5a receptor antagonist avacopan for severe granulomatosis with polyangiitis (GPA) and microscopic polyangiitis (MPA), and antiinterleukin-5 therapies in the management of eosinophilic granulomatosis with polyangiitis (EGPA). Avacopan was approved by the US Food and Drug Administration (FDA) and European Medicines Agency (EMA) for the treatment of AAV in 2021, following the phase 3 ADVOCAT E trial comparing oral avacopan to an oral GC taper for GPA and MPA.
View Article and Find Full Text PDFSemin Nephrol
March 2025
Department of Pathology and Laboratory Medicine, Cedars-Sinai Medical Center, Los Angeles, CA 90048 USA. Electronic address:
IgA nephropathy (IgAN), the world's most common form of primary glomerulonephritis (GN), has a variable clinical and pathologic presentation. While all cases of IgAN show dominant or codominant glomerular IgA deposits, their histologic appearance can range from essentially normal to severe crescentic GN. Oxford (MEST-C) scoring is widely used to classify IgAN on kidney biopsies and has been validated to correlate with clinical presentation and as an independent predictor of kidney outcomes in multiple studies.
View Article and Find Full Text PDFEnter search terms and have AI summaries delivered each week - change queries or unsubscribe any time!