Childhood Kidney Diseases

Korean Society of Pediatric Nephrology (대한소아신장학회)
권호 표지
DOI QR코드

DOI QR Code

Anti-glomerular basement membrane disease with concomitant myeloperoxidase antibodies in a pediatric patient: a case report of presentation, treatment and outcome a rare disease

  • Yiressy Pina (Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine) ;
  • Emily Frierson (Division of Nephrology, Department of Pediatrics, University of South Florida (USF) Health Morsani College School of Medicine) ;
  • John Hicks (Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine) ;
  • Partha Chakraborty (Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine) ;
  • Alvaro Orjuela (Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine) ;
  • Amit Thakral (Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine)
  • Received : 2025.05.02
  • Accepted : 2025.07.04
  • Published : 2026.02.28
Download PDF
⟨ Previous Next ⟩

Abstract

Anti-glomerular basement membrane disease (anti-GBMD) is a rare small-vessel vasculitis presenting as rapidly progressive glomerulonephritis and/or diffuse alveolar hemorrhage. A third of patients with anti-GBMD have antineutrophil cytoplasmic antibodies and are known as double-positive patients. There are a limited number of pediatric double-positive cases published in the literature. In this case report, we summarize the presentation, treatment, and outcome of a patient with anti-GBMD with both anti-myeloperoxidase and anti-GBM antibodies, highlighting the safe use of rituximab. We also describe atypical complications in our case, including significant weight loss of unclear etiology and development of recurrent episodes of acute pericalcific arthritis.

Keywords

References

  1. Bharati J, Jhaveri KD, Salama AD, Oni L. Anti-glomerular basement membrane disease: recent updates. Adv Kidney Dis Health 2024;31:206-15. https://doi.org/10.1053/j.akdh.2024.04.007
  2. Kuang H, Jiang N, Jia XY, Cui Z, Zhao MH. Epidemiology, clinical features, risk factors, and outcomes in anti-glomerular basement membrane disease: a systematic review and meta-analysis. Autoimmun Rev 2024;23:103531. https://doi.org/10.1016/j.autrev.2024.103531
  3. Philip R, Dumont A, Martin Silva N, de Boysson H, Aouba A, Deshayes S. ANCA and anti-glomerular basement membrane double-positive patients: a systematic review of the literature. Autoimmun Rev 2021;20:102885. https://doi.org/10.1016/j.autrev.2021.102885
  4. Kidney Disease: Improving Global Outcomes (KDIGO) Glomerular Diseases Work Group. KDIGO 2021 clinical practice guideline for the management of glomerular diseases. Kidney Int 2021;100:S1-S276. https://doi.org/10.1016/j.kint.2021.05.021
  5. Bayat A, Kamperis K, Herlin T. Characteristics and outcome of Goodpasture's disease in children. Clin Rheumatol 2012;31:1745-51. https://doi.org/10.1007/s10067-012-2062-9
  6. Williamson SR, Phillips CL, Andreoli SP, Nailescu C. A 25-year experience with pediatric anti-glomerular basement membrane disease. Pediatr Nephrol 2011;26:85-91. https://doi.org/10.1007/s00467-010-1663-2
  7. Jayne DR, Marshall PD, Jones SJ, Lockwood CM. Autoantibodies to GBM and neutrophil cytoplasm in rapidly progressive glomerulonephritis. Kidney Int 1990;37:965-70. https://doi.org/10.1038/ki.1990.72
  8. Echcharii N, Essadiqi S, Chekhlabi N, Tissir R, Hadri H, Allaoui A, et al. Pediatric double-seropositive anti-glomerular basement membrane antibody disease: a case report and literature review. Clin Nephrol Case Stud 2024;12:60-72. https://doi.org/10.5414/CNCS111439
  9. Helander L, Hanna M, Annen K. Pediatric double positive anti-glomerular basement membrane antibody and anti-neutrophil cytoplasmic antibody glomerulonephritis: a case report with review of literature. J Clin Apher 2021;36:505-10. https://doi.org/10.1002/jca.v36.3
  10. Timmermans SAMEG, van Dam MJCM, Vink E, Horuz FAPT, van Paassen P, Rosias PPR. Rituximab for the treatment of pediatric double-positive small-vessel vasculitis. Kidney Int Rep 2020;5:235-8. https://doi.org/10.1016/j.ekir.2019.11.009
  11. Agarwal I, Al-Ghitany A. Anti-glomerular basement membrane: a rare cause of renal failure in children. Saudi J Kidney Dis Transpl 2017;28:379-83. https://doi.org/10.4103/1319-2442.202780
  12. Xie LJ, Cui Z, Jia XY, Chen Z, Liu XR, Zhao MH. Coexistence of anti-glomerular basement membrane antibodies and anti-neutrophil cytoplasmic antibodies in a child with human leukocyte antigen susceptibility and detailed antibody description: a case report. Medicine (Baltimore) 2015;94:e1179.
  13. Bogdanovic R, Minic P, Markovic-Lipkovski J, Stajic N, Savic N, Rodic M. Pulmonary renal syndrome in a child with coexistence of anti-neutrophil cytoplasmic antibodies and anti-glomerular basement membrane disease: case report and literature review. BMC Nephrol 2013;14:66. https://doi.org/10.1186/1471-2369-14-66
  14. Poddar B, Singhal S, Azim A, Gulati S, Baronia A. Goodpasture's syndrome in children. Saudi J Kidney Dis Transpl 2010;21:935-9.
  15. Naidoo S, Waller S. Anti-GBM antibodies co-exist with MPO-ANCA in a 4-year-old girl with acute renal failure. Pediatr Nephrol 2009;24:215-6. https://doi.org/10.1007/s00467-008-0928-5
  16. Hijosa MM, Roman LE, Camblor CF, Garcia ML, Salcedo DP, Torres MN. Anti-GBM and anti-MPO antibodies coexist in a case of pulmonary renal syndrome. Pediatr Nephrol 2005;20:807-10. https://doi.org/10.1007/s00467-004-1762-z
  17. Paueksakon P, Hunley TE, Lee SM, Fogo AB. A 12-year-old girl with pulmonary hemorrhage, skin lesions, and hematuria. Am J Kidney Dis 1999;33:404-9. https://doi.org/10.1016/S0272-6386(99)70321-5
  18. Fanburg BL, Niles JL, Mark EJ. Case 52-1993: a 17-year-old girl with massive hemoptysis and acute oliguric renal failure. N Engl J Med 1993;329:2019-26. https://doi.org/10.1056/NEJM199312303292708
  19. Connelly-Smith L, Alquist CR, Aqui NA, Hofmann JC, Klingel R, Onwuemene OA, et al. Guidelines on the use of therapeutic apheresis in clinical practice–evidence-based approach from the Writing Committee of the American Society for Apheresis: the ninth special issue. J Clin Apher 2023;38:77-278.
  20. Doumas C, Vazirani RM, Clifford PD, Owens P. Acute calcific periarthritis of the hand and wrist: a series and review of the literature. Emerg Radiol 2007;14:199-203. https://doi.org/10.1007/s10140-007-0626-9