atypical hemolytic uremic syndrome (aHUS) complement copy number variations (CNVs) eculizumab factor H (FH) factor H-related proteins (FHRs) single molecule real-time (SMRT) structural variants (SVs)

Journal

Frontiers in immunology
ISSN: 1664-3224
Titre abrégé: Front Immunol
Pays: Switzerland
ID NLM: 101560960

Informations de publication

Date de publication:
2022
Historique:
received: 04 08 2022
accepted: 28 12 2022
entrez: 16 2 2023
pubmed: 17 2 2023
medline: 18 2 2023
Statut: epublish

Résumé

Atypical hemolytic uremic syndrome (aHUS) is a rare disease that manifests with microangiopathic hemolytic anemia, thrombocytopenia, and acute renal failure, and is associated with dysregulation of the alternative complement pathway. The chromosomal region including In this study, we report the results of We found uncommon SVs in 8% of patients with primary aHUS: 70% carried rearrangements involving In conclusion, these data highlight that uncommon

Identifiants

pubmed: 36793547
doi: 10.3389/fimmu.2022.1011580
pmc: PMC9923232
doi:

Substances chimiques

Complement Factor H 80295-65-4

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

1011580

Informations de copyright

Copyright © 2023 Piras, Valoti, Alberti, Bresin, Mele, Breno, Liguori, Donadelli, Rigoldi, Benigni, Remuzzi and Noris.

Déclaration de conflit d'intérêts

MN has received honoraria from Alexion Pharmaceuticals for giving lectures, and for participating in advisory boards, and she has received research grants from Omeros, Gemini, Novartis and BioCryst Pharmaceuticals. AB has received honoraria from Alexion Phamaceuticals and BioCryst Pharmaceuticals. GR has consultancy agreements with AbbVie, Alexion Pharmaceuticals, Novartis Pharma and BioCryst Pharmaceuticals. Since 1st May 2022, the co-author EV has been employed by Frontiers Media SA. EV declared her affiliation with Frontiers, and the handling Editor states that the process nevertheless met the standards of a fair and objective review. The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be constructed as a potential conflict of interest.

Références

Br J Pharmacol. 2021 Jul;178(14):2849-2862
pubmed: 32585035
Pediatr Nephrol. 2013 Nov;28(11):2221-5
pubmed: 23880784
Nat Rev Genet. 2007 Oct;8(10):762-75
pubmed: 17846636
Front Immunol. 2019 May 01;10:853
pubmed: 31118930
Clin J Am Soc Nephrol. 2010 Oct;5(10):1844-59
pubmed: 20595690
Nephrol Dial Transplant. 2017 Mar 1;32(3):466-474
pubmed: 28339660
Immunopharmacology. 1999 May;42(1-3):53-60
pubmed: 10408366
Trends Immunol. 2008 Aug;29(8):380-7
pubmed: 18602340
J Am Soc Nephrol. 2013 Feb;24(3):475-86
pubmed: 23431077
Trends Immunol. 2015 Jun;36(6):374-84
pubmed: 25979655
Proc Natl Acad Sci U S A. 2013 Mar 19;110(12):4685-90
pubmed: 23487775
J Am Soc Nephrol. 2019 Dec;30(12):2449-2463
pubmed: 31575699
J Med Genet. 2005 Nov;42(11):852-6
pubmed: 15784724
Nat Genet. 2010 Jul;42(7):553-5; author reply 555-6
pubmed: 20581873
Hum Mutat. 2006 Sep;27(9):938-45
pubmed: 16917894
Front Med (Lausanne). 2020 Nov 03;7:579418
pubmed: 33224962
J Am Soc Nephrol. 2015 Jan;26(1):209-19
pubmed: 24904082
Nat Genet. 2013 May;45(5):531-6
pubmed: 23542698
J Clin Invest. 2013 Jun;123(6):2434-46
pubmed: 23728178
Immunol Rev. 2023 Jan;313(1):25-45
pubmed: 36382387
Kidney Int. 2017 Mar;91(3):539-551
pubmed: 27989322
Immunogenetics. 1999 Jun;49(6):549-52
pubmed: 10380701
Clin J Am Soc Nephrol. 2015 Jun 5;10(6):1011-9
pubmed: 25854283
Hum Mutat. 1992;1(1):3-12
pubmed: 1301188
J Am Soc Nephrol. 2020 Feb;31(2):241-256
pubmed: 31980588
J Am Soc Nephrol. 2005 Apr;16(4):1035-50
pubmed: 15728781
Transplant Proc. 2020 Jan - Feb;52(1):146-152
pubmed: 31924403
PLoS Med. 2006 Oct;3(10):e431
pubmed: 17076561
Nephron. 2019;142(3):264-270
pubmed: 30889567
Hum Mol Genet. 2003 Dec 15;12(24):3385-95
pubmed: 14583443
Blood. 2006 Aug 15;108(4):1267-79
pubmed: 16621965
Eur J Hum Genet. 2005 Nov;13(11):1231-4
pubmed: 16030524
Am J Hum Genet. 2002 Dec;71(6):1285-95
pubmed: 12424708
J Am Soc Nephrol. 2016 May;27(5):1305-11
pubmed: 26376859
J Am Soc Nephrol. 2018 Jan;29(1):240-249
pubmed: 28993505
Mol Immunol. 2015 Oct;67(2 Pt B):276-86
pubmed: 26163426
Front Immunol. 2017 Oct 18;8:1328
pubmed: 29093712
Mol Immunol. 2013 Dec 15;56(3):170-80
pubmed: 23830046
Nephrol Dial Transplant. 2011 Feb;26(2):739-41
pubmed: 20974643
Hum Mutat. 2006 Mar;27(3):292-3
pubmed: 16470555
Blood. 2010 Jan 14;115(2):379-87
pubmed: 19861685
Front Genet. 2021 Jun 11;12:670727
pubmed: 34211499
J Hum Hypertens. 2000 Oct-Nov;14(10-11):687-90
pubmed: 11095159
Blood. 2020 Sep 24;136(13):1558-1561
pubmed: 32488248
Blood. 2021 Jun 24;137(25):3484-3494
pubmed: 33651882
Vaccine. 2008 Dec 30;26 Suppl 8:I9-14
pubmed: 19388158
Front Immunol. 2021 Feb 25;12:615748
pubmed: 33732239
Hum Mol Genet. 2005 Mar 1;14(5):703-12
pubmed: 15661753

Auteurs

Rossella Piras (R)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Elisabetta Valoti (E)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Marta Alberti (M)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Elena Bresin (E)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Caterina Mele (C)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Matteo Breno (M)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Lucia Liguori (L)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Roberta Donadelli (R)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Miriam Rigoldi (M)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Ariela Benigni (A)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Giuseppe Remuzzi (G)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

Marina Noris (M)

Istituto di Ricerche Farmacologiche Mario Negri IRCCS, Clinical Research Center for Rare Diseases Aldo e Cele Daccò and Centro Anna Maria Astori, Science and Technology Park Kilometro Rosso, Bergamo, Italy.

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Classifications MeSH