References

Key articles

Brodszki N, Frazer-Abel A, Grumach AS, et al. European Society for Immunodeficiencies (ESID) and European Reference Network on Rare Primary Immunodeficiency, Autoinflammatory and Autoimmune Diseases (ERN RITA) complement guideline: deficiencies, diagnosis, and management. J Clin Immunol. 2020 May;40(4):576-91.Full text  Abstract

Grumach AS, Kirschfink M. Are complement deficiencies really rare? Overview on prevalence, clinical importance and modern diagnostic approach. Mol Immunol. 2014 Oct;61(2):110-7. Abstract

Maurer M, Magerl M, Betschel S, et al. The international WAO/EAACI guideline for the management of hereditary angioedema-the 2021 revision and update. Allergy. 2022 Jul;77(7):1961-90.Full text  Abstract

Reference articles

1. Mastellos DC, Hajishengallis G, Lambris JD. A guide to complement biology, pathology and therapeutic opportunity. Nat Rev Immunol. 2024 Feb;24(2):118-41. Abstract

2. Jayaraman A, Walachowski S, Bosmann M. The complement system: a key player in the host response to infections. Eur J Immunol. 2024 Nov;54(11):e2350814.Full text  Abstract

3. Hajishengallis G, Reis ES, Mastellos DC, et al. Novel mechanisms and functions of complement. Nat Immunol. 2017 Nov 16;18(12):1288-8. Abstract

4. Reis ES, Mastellos DC, Hajishengallis G, et al. New insights into the immune functions of complement. Nat Rev Immunol. 2019 Aug;19(8):503-16.Full text  Abstract

5. Ekdahl KN, Teramura Y, Hamad OA, et al. Dangerous liaisons: complement, coagulation, and kallikrein/kinin cross-talk act as a linchpin in the events leading to thromboinflammation. Immunol Rev. 2016 Nov;274(1):245-69. Abstract

6. Bekassy Z, Lopatko Fagerström I, Bader M, et al. Crosstalk between the renin-angiotensin, complement and kallikrein-kinin systems in inflammation. Nat Rev Immunol. 2022 Jul;22(7):411-28.Full text  Abstract

7. Zipfel PF, Skerka C. Complement regulators and inhibitory proteins. Nat Rev Immunol. 2009 Oct;9(10):729-40. Abstract

8. Ricklin D, Lambris JD. Complement in immune and inflammatory disorders: pathophysiological mechanisms. J Immunol. 2013 Apr 15;190(8):3831-8.Full text  Abstract

9. Karasu E, Nilsson B, Köhl J, et al. Targeting complement pathways in polytrauma- and sepsis-induced multiple-organ dysfunction. Front Immunol. 2019;10:543.Full text  Abstract

10. Koski CL, Sanders ME, Swoveland PT, et al. Activation of terminal components of complement in patients with Guillain-Barré syndrome and other demyelinating neuropathies. J Clin Invest. 1987 Nov;80(5):1492-7.Full text  Abstract

11. Saez-Calveras N, Stuve O. The role of the complement system in multiple sclerosis: a review. Front Immunol. 2022 Aug 10;13:970486.Full text  Abstract

12. Bohlson SS, Tenner AJ. Complement in the brain: contributions to neuroprotection, neuronal plasticity, and neuroinflammation. Annu Rev Immunol. 2023 Apr 26;41:431-52.Full text  Abstract

13. Tenner AJ, Petrisko TJ. Knowing the enemy: strategic targeting of complement to treat Alzheimer disease. Nat Rev Neurol. 2025 May;21(5):250-64.Full text  Abstract

14. Ekdahl KN, Huang S, Nilsson B, et al. Complement inhibition in biomaterial- and biosurface-induced thromboinflammation. Semin Immunol. 2016 Jun;28(3):268-77.Full text  Abstract

15. Brodszki N, Frazer-Abel A, Grumach AS, et al. European Society for Immunodeficiencies (ESID) and European Reference Network on Rare Primary Immunodeficiency, Autoinflammatory and Autoimmune Diseases (ERN RITA) complement guideline: deficiencies, diagnosis, and management. J Clin Immunol. 2020 May;40(4):576-91.Full text  Abstract

16. Degn SE, Jensenius JC, Thiel S. Disease-causing mutations in genes of the complement system. Am J Hum Genet. 2011 Jun 10;88(6):689-705.Full text  Abstract

17. Skattum L, Van deuren M, Van der poll T, et al. Complement deficiency states and associated infections. Mol Immunol. 2011 Aug;48(14):1643-55. Abstract

18. Figueroa JE, Densen P. Infectious diseases associated with complement deficiencies. Clin Microbiol Rev. 1991 Jul;4(3):359-95.Full text  Abstract

19. Schröder-Braunstein J, Kirschfink M. Complement deficiencies and dysregulation: pathophysiological consequences, modern analysis, and clinical management. Mol Immunol. 2019 Oct;114:299-311. Abstract

20. West EE, Kemper C. Complosome - the intracellular complement system. Nat Rev Nephrol. 2023 Jul;19(7):426-39.Full text  Abstract

21. King BC, Blom AM. Intracellular complement and immunometabolism: the advantages of compartmentalization. Eur J Immunol. 2024 Aug;54(8):e2350813.Full text  Abstract

22. Mayilyan KR. Complement genetics, deficiencies, and disease associations. Protein Cell. 2012 Jul;3(7):487-96.Full text  Abstract

23. Merle NS, Church SE, Fremeaux-Bacchi V, et al. Complement system part I - molecular mechanisms of activation and regulation. Front Immunol. 2015 Jun 2;6:262.Full text  Abstract

24. Bohlson SS, Garred P, Kemper C, et al. Complement nomenclature-deconvoluted. Front Immunol. 2019;10:1308.Full text  Abstract

25. Noris M, Remuzzi G. Overview of complement activation and regulation. Semin Nephrol. 2013 Nov;33(6):479-92.Full text  Abstract

26. Lhotta K, Janecke AR, Scheiring J, et al. A large family with a gain-of-function mutation of complement C3 predisposing to atypical hemolytic uremic syndrome, microhematuria, hypertension and chronic renal failure. Clin J Am Soc Nephrol. 2009 Aug;4(8):1356-62.Full text  Abstract

27. Urban A, Volokhina E, Felberg A, et al. Gain-of-function mutation in complement C2 protein identified in a patient with aHUS. J Allergy Clin Immunol. 2020 Oct;146(4):916-9.e11.Full text  Abstract

28. Fakhouri F, Frémeaux-Bacchi V. Thrombotic microangiopathy in aHUS and beyond: clinical clues from complement genetics. Nat Rev Nephrol. 2021 Aug;17(8):543-53. Abstract

29. Grumach AS, Kirschfink M. Are complement deficiencies really rare? Overview on prevalence, clinical importance and modern diagnostic approach. Mol Immunol. 2014 Oct;61(2):110-7. Abstract

30. Kindle G, Alligon M, Albert MH, et al. Inborn errors of immunity: manifestation, treatment, and outcome - an ESID registry 1994-2024 report on 30,628 patients. medRxiv. 2025 Apr 16:2025.02.20.25322586.Full text  Abstract

31. Kim VHD, Upton JEM, Derfalvi B, et al. Inborn errors of immunity (primary immunodeficiencies). Allergy Asthma Clin Immunol. 2025 Jan 8;20(suppl 3):76.Full text  Abstract

32. Unsworth DJ. Complement deficiency and disease. J Clin Pathol. 2008 Sep;61(9):1013-7. Abstract

33. Ellison RT 3rd, Kohler PF, Curd JG, et al. Prevalence of congenital or acquired complement deficiency in patients with sporadic meningococcal disease. N Engl J Med. 1983 Apr 21;308(16):913-6. Abstract

34. Tangye SG, Al-Herz W, Bousfiha A, et al. Human inborn errors of immunity: 2022 update on the classification from the International Union of Immunological Societies Expert Committee. J Clin Immunol. 2022 Oct;42(7):1473-507.Full text  Abstract

35. Maurer M, Magerl M, Betschel S, et al. The international WAO/EAACI guideline for the management of hereditary angioedema-the 2021 revision and update. Allergy. 2022 Jul;77(7):1961-90.Full text  Abstract

36. Levy JH, Freiberger DJ, Roback J. Hereditary angioedema: current and emerging treatment options. Anesth Analg. 2010 May 1;110(5):1271-80.Full text  Abstract

37. Caccia S, Suffritti C, Cicardi M. Pathophysiology of hereditary angioedema. Pediatr Allergy Immunol Pulmonol. 2014 Dec 1;27(4):159-63.Full text  Abstract

38. Grumach AS, Riedl MA, Cheng L, et al. Hereditary angioedema diagnosis: reflecting on the past, envisioning the future. World Allergy Organ J. 2025 Jun;18(6):101060.Full text  Abstract

39. Bork K, Staubach-Renz P, Hardt J. Angioedema due to acquired C1-inhibitor deficiency: spectrum and treatment with C1-inhibitor concentrate. Orphanet J Rare Dis. 2019 Mar 13;14(1):65.Full text  Abstract

40. Shi Y, Wang C. Where we are with acquired angioedema due to C1 inhibitor deficiency: a systematic literature review. Clin Immunol. 2021 Sep;230:108819. Abstract

41. Zuraw BL, Bork K, Bouillet L, et al. Hereditary angioedema with normal C1 inhibitor: an updated International Consensus Paper on diagnosis, pathophysiology, and treatment. Clin Rev Allergy Immunol. 2025 Mar 7;68(1):24.Full text  Abstract

42. Wang RX, Newman SA. Urticarial vasculitis. Immunol Allergy Clin North Am. 2024 Aug;44(3):483-502. Abstract

43. Macedo AC, Isaac L. Systemic lupus erythematosus and deficiencies of early components of the complement classical pathway. Front Immunol. 2016 Feb 24;7:55.Full text  Abstract

44. Coss SL, Zhou D, Chua GT, et al. The complement system and human autoimmune diseases. J Autoimmun. 2023 May;137:102979.Full text  Abstract

45. Trendelenburg M. Autoantibodies against complement component C1q in systemic lupus erythematosus. Clin Transl Immunology. 2021 Apr 29;10(4):e1279.Full text  Abstract

46. Carroll MC. A protective role for innate immunity in systemic lupus erythematosus. Nat Rev Immunol. 2004 Oct;4(10):825-31. Abstract

47. Buso H, Triaille C, Flinn AM, et al. Update on hereditary C1q deficiency: pathophysiology, clinical presentation, genotype and management. Curr Opin Allergy Clin Immunol. 2024 Dec 1;24(6):427-33. Abstract

48. Weinstein A, Alexander RV, Zack DJ. A review of complement activation in SLE. Curr Rheumatol Rep. 2021 Feb 10;23(3):16.Full text  Abstract

49. Lewis LA, Ram S. Meningococcal disease and the complement system. Virulence. 2014 Jan 1;5(1):98-126.Full text  Abstract

50. UK Health Security Agency. The complete routine immunisation schedule from February 2022. Feb 2022 [internet publication].Full text

51. Centers for Disease Control and Prevention. Child and adolescent immunization schedule by age: recommendations for ages 18 years or younger, United States, 2025. Oct 2025 [internet publication].Full text

52. Centers for Disease Control and Prevention. Adult immunization schedule by age: recommendations for ages 19 years or older, United States, 2025. Oct 2025 [internet publication].Full text

53. Noris M, Remuzzi G. Genetics of immune-mediated glomerular diseases: focus on complement. Semin Nephrol. 2017 Sep;37(5):447-63. Abstract

54. Willows J, Brown M, Sheerin NS. The role of complement in kidney disease. Clin Med (Lond). 2020 Mar;20(2):156-60.Full text  Abstract

55. Petr V, Thurman JM. The role of complement in kidney disease. Nat Rev Nephrol. 2023 Dec;19(12):771-87. Abstract

56. Smith RJH, Appel GB, Blom AM, et al. C3 glomerulopathy - understanding a rare complement-driven renal disease. Nat Rev Nephrol. 2019 Mar;15(3):129-43.Full text  Abstract

57. Wooden B, Nester CM, Bomback AS. Update on C3 glomerulopathy. Adv Kidney Dis Health. 2024 May;31(3):223-33. Abstract

58. Cook HT. C3 glomerulopathy. F1000Res. 2017 Mar 10;6:248.Full text  Abstract

59. Corvillo F, Okrój M, Nozal P, et al. Nephritic factors: an overview of classification, diagnostic tools and clinical associations. Front Immunol. 2019 Apr 24;10:886.Full text  Abstract

60. Welsh SJ, Zhang Y, Smith RJH. Acquired drivers of C3 glomerulopathy. Clin Kidney J. 2025 Mar;18(3):sfaf022.Full text  Abstract

61. Donadelli R, Sinha A, Bagga A, et al. HUS and TTP: traversing the disease and the age spectrum. Semin Nephrol. 2023 Jul;43(4):151436. Abstract

62. Jokiranta TS. HUS and atypical HUS. Blood. 2017 May 25;129(21):2847-56.Full text  Abstract

63. Wilke GA, Apte RS. Complement regulation in the eye: implications for age-related macular degeneration. J Clin Invest. 2024 May 1;134(9):e178296.Full text  Abstract

64. Fritsche LG, Igl W, Bailey JN, et al. A large genome-wide association study of age-related macular degeneration highlights contributions of rare and common variants. Nat Genet. 2016 Feb;48(2):134-43.Full text  Abstract

65. Hill A, DeZern AE, Kinoshita T, et al. Paroxysmal nocturnal haemoglobinuria. Nat Rev Dis Primers. 2017 May 18;3:17028. Abstract

66. Risitano AM, Frieri C, Urciuoli E, et al. The complement alternative pathway in paroxysmal nocturnal hemoglobinuria: from a pathogenic mechanism to a therapeutic target. Immunol Rev. 2023 Jan;313(1):262-78.Full text  Abstract

67. Ozen A, Comrie WA, Ardy RC, et al. CD55 deficiency, early-onset protein-losing enteropathy, and thrombosis. N Engl J Med. 2017 Jul 6;377(1):52-61.Full text  Abstract

68. Almutawea LM, Hajeri AAA, Farid EM, et al. Inherited CD59 deficiency, where neurology and genetics intertwine. Neurosciences (Riyadh). 2023 Apr;28(2):130-5.Full text  Abstract

69. Gorevic PD. Rheumatoid factor, complement, and mixed cryoglobulinemia. Clin Dev Immunol. 2012;2012:439018.Full text  Abstract

70. Levy Y, George J, Yona E, et al. Partial lipodystrophy, mesangiocapillary glomerulonephritis, and complement dysregulation. An autoimmune phenomenon. Immunol Res. 1998 Aug;18(1):55-60. Abstract

71. Corvillo F, Akinci B. An overview of lipodystrophy and the role of the complement system. Mol Immunol. 2019 Aug;112:223-32. Abstract

72. Prohászka Z, Nilsson B, Frazer-Abel A, et al. Complement analysis 2016: clinical indications, laboratory diagnostics and quality control. Immunobiology. 2016 Nov;221(11):1247-58. Abstract

73. Ling M, Murali M. Analysis of the complement system in the clinical immunology laboratory. Clin Lab Med. 2019 Dec;39(4):579-90. Abstract

74. Frazer-Abel A, Kirschfink M, Prohászka Z. Expanding horizons in complement analysis and quality control. Front Immunol. 2021 Aug 9;12:697313.Full text  Abstract

75. Seelen MA, Roos A, Wieslander J, et al. Functional analysis of the classical, alternative, and MBL pathways of the complement system: standardization and validation of a simple ELISA. J Immunol Methods. 2005 Jan;296(1-2):187-98. Abstract

76. Wehling C, Amon O, Bommer M, et al. Monitoring of complement activation biomarkers and eculizumab in complement-mediated renal disorders. Clin Exp Immunol. 2017 Feb;187(2):304-15.Full text  Abstract

77. Germenis AE, Margaglione M, Pesquero JB, et al. International consensus on the use of genetics in the management of hereditary angioedema. J Allergy Clin Immunol Pract. 2020 Mar;8(3):901-11. Abstract

78. Licht C, Weyersburg A, Heinen S, et al. Successful plasma therapy for atypical hemolytic uremic syndrome caused by factor H deficiency owing to a novel mutation in the complement cofactor protein domain 15. Am J Kidney Dis. 2005 Feb;45(2):415-21. Abstract

79. Ricklin D, Mastellos DC, Reis ES, et al. The renaissance of complement therapeutics. Nat Rev Nephrol. 2018 Jan;14(1):26-47.Full text  Abstract

80. Mastellos DC, Ricklin D, Lambris JD. Clinical promise of next-generation complement therapeutics. Nat Rev Drug Discov. 2019 Sep;18(9):707-29.Full text  Abstract

81. Ricklin D. Complement-targeted therapeutics: are we there yet, or just getting started? Eur J Immunol. 2024 Dec;54(12):e2350816.Full text  Abstract

82. West EE, Woodruff T, Fremeaux-Bacchi V, et al. Complement in human disease: approved and up-and-coming therapeutics. Lancet. 2024 Jan 27;403(10424):392-405.Full text  Abstract

83. Greve J, Lochbaum R, Trainotti S, et al. The international HAE guideline under real-life conditions: from possibilities to limits in daily life - current real-world data of 8 German angioedema centers. Allergol Select. 2024;8:346-57.Full text  Abstract

84. Betschel SD, Banerji A, Busse PJ, et al. Hereditary angioedema: a review of the current and evolving treatment landscape. J Allergy Clin Immunol Pract. 2023 Aug;11(8):2315-25. Abstract

85. Aygören-Pürsün E, Zanichelli A, Cohn DM, et al. An investigational oral plasma kallikrein inhibitor for on-demand treatment of hereditary angioedema: a two-part, randomised, double-blind, placebo-controlled, crossover phase 2 trial. Lancet. 2023 Feb 11;401(10375):458-69. Abstract

86. Centers for Disease Control and Prevention. Vaccine recommendations and guidelines of the ACIP. General best practice guidelines for immunization: altered immunocompetence. Aug 2023 [internet publication].Full text

87. Caballero T, Farkas H, Bouillet L, et al. International consensus and practical guidelines on the gynecologic and obstetric management of female patients with hereditary angioedema caused by C1 inhibitor deficiency. J Allergy Clin Immunol. 2012 Feb;129(2):308-20.Full text  Abstract

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