Complement Activation and Thrombotic Microangiopathies

Background and objectivesAtypical hemolytic uremic syndrome is a form of thrombotic microangiopathy caused by dysregulation of the alternative complement pathway. There is evidence showing complement activation in other thrombotic microangiopathies. The aim of this study was to evaluate complement a...

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Autores: Palomo M, Blasco M, Molina P, Lozano M, Praga M, Torramade-Moix S, Martinez-Sanchez J, Cid J, Escolar G, Carreras E, Paules C, Crispi F, Quintana LF, Poch E, Rodas L, Goma E, Morelle J, Espinosa M, Morales E, Avila A, Cabello V, Ariceta G, Chocron S, Manrique J, Barros X, Martin N, Huerta A, Fraga-Rodriguez GM, Cao M, Martin M, Romera AM, Moreso F, Manonelles A, Gratacos E, Pereira A, Campistol JM, Diaz-Ricart M
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2019
País:España
Institución:Fundación para el Fomento de la Investigación Sanitaria y Biomédica de la Comunitat Valenciana (FISABIO)
Repositorio:r-FISABIO. Repositorio Institucional de Producción Científica
OAI Identifier:oai:fisabio.fundanetsuite.com:p4518
Acceso en línea:https://fisabio.portalinvestigacion.com/publicaciones/4518
Access Level:acceso abierto
Palabra clave:humans
pregnancy
female
atypical hemolytic uremic syndrome
complement membrane
HELLP syndrome
complement C9
eculizumab
complement pathway
alternative
pre-eclampsia
hypertension
malignant
fibrin
antibodies
monoclonal
humanized
complement system proteins
complement activation
thrombotic microangiopathies
recurrence
fluorescent antibody technique
endothelial cells
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spelling Complement Activation and Thrombotic MicroangiopathiesPalomo MBlasco MMolina PLozano MPraga MTorramade-Moix SMartinez-Sanchez JCid JEscolar GCarreras EPaules CCrispi FQuintana LFPoch ERodas LGoma EMorelle JEspinosa MMorales EAvila ACabello VAriceta GChocron SManrique JBarros XMartin NHuerta AFraga-Rodriguez GMCao MMartin MRomera AMMoreso FManonelles AGratacos EPereira ACampistol JMDiaz-Ricart Mhumanspregnancyfemaleatypical hemolytic uremic syndromecomplement membraneHELLP syndromecomplement C9eculizumabcomplement pathwayalternativepre-eclampsiahypertensionmalignantfibrinantibodiesmonoclonalhumanizedcomplement system proteinscomplement activationthrombotic microangiopathiesrecurrencefluorescent antibody techniqueendothelial cellsBackground and objectivesAtypical hemolytic uremic syndrome is a form of thrombotic microangiopathy caused by dysregulation of the alternative complement pathway. There is evidence showing complement activation in other thrombotic microangiopathies. The aim of this study was to evaluate complement activation in different thrombotic microangiopathies and to monitor treatment response.Design, setting, participants, & measurementsComplement activation was assessed by exposing endothelial cells to sera or activated-patient plasma?citrated plasma mixed with a control sera pool (1:1)?to analyze C5b-9 deposits by immunofluorescence. Patients with atypical hemolytic uremic syndrome (n=34) at different stages of the disease, HELLP syndrome (a pregnancy complication characterized by hemolysis, elevated liver enzymes, and low platelet count) or severe preeclampsia (n=10), and malignant hypertension (n=5) were included.ResultsAcute phase atypical hemolytic uremic syndrome?activated plasma induced an increased C5b-9 deposition on endothelial cells. Standard and lower doses of eculizumab inhibited C5b-9 deposition in all patients with atypical hemolytic uremic syndrome, except in two who showed partial remission and clinical relapse. Significant fibrin formation was observed together with C5b-9 deposition. Results obtained using activated-plasma samples were more marked and reproducible than those obtained with sera. C5b-9 deposition was also increased with samples from patients with HELLP (all cases) and preeclampsia (90%) at disease onset. This increase was sustained in those with HELLP after 40 days, and levels normalized in patients with both HELLP and preeclampsia after 6?9 months. Complement activation in those with malignant hypertension was at control levels.ConclusionsThe proposed methodology identifies complement overactivation in patients with atypical hemolytic uremic syndrome at acute phase and in other diseases such as HELLP syndrome and preeclampsia. Moreover, it is sensitive enough to individually assess the efficiency of the C5 inhibition treatment.American Society of Nephrology2019info:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersionhttps://fisabio.portalinvestigacion.com/publicaciones/4518Clinical journal of the American Society of NephrologyISSN: 1555905XISSNe: 15559041reponame:r-FISABIO. Repositorio Institucional de Producción Científicainstname:Fundación para el Fomento de la Investigación Sanitaria y Biomédica de la Comunitat Valenciana (FISABIO)Inglésinfo:eu-repo/semantics/openAccessoai:fisabio.fundanetsuite.com:p45182026-06-11T12:45:17Z
dc.title.none.fl_str_mv Complement Activation and Thrombotic Microangiopathies
title Complement Activation and Thrombotic Microangiopathies
spellingShingle Complement Activation and Thrombotic Microangiopathies
Palomo M
humans
pregnancy
female
atypical hemolytic uremic syndrome
complement membrane
HELLP syndrome
complement C9
eculizumab
complement pathway
alternative
pre-eclampsia
hypertension
malignant
fibrin
antibodies
monoclonal
humanized
complement system proteins
complement activation
thrombotic microangiopathies
recurrence
fluorescent antibody technique
endothelial cells
title_short Complement Activation and Thrombotic Microangiopathies
title_full Complement Activation and Thrombotic Microangiopathies
title_fullStr Complement Activation and Thrombotic Microangiopathies
title_full_unstemmed Complement Activation and Thrombotic Microangiopathies
title_sort Complement Activation and Thrombotic Microangiopathies
dc.creator.none.fl_str_mv Palomo M
Blasco M
Molina P
Lozano M
Praga M
Torramade-Moix S
Martinez-Sanchez J
Cid J
Escolar G
Carreras E
Paules C
Crispi F
Quintana LF
Poch E
Rodas L
Goma E
Morelle J
Espinosa M
Morales E
Avila A
Cabello V
Ariceta G
Chocron S
Manrique J
Barros X
Martin N
Huerta A
Fraga-Rodriguez GM
Cao M
Martin M
Romera AM
Moreso F
Manonelles A
Gratacos E
Pereira A
Campistol JM
Diaz-Ricart M
author Palomo M
author_facet Palomo M
Blasco M
Molina P
Lozano M
Praga M
Torramade-Moix S
Martinez-Sanchez J
Cid J
Escolar G
Carreras E
Paules C
Crispi F
Quintana LF
Poch E
Rodas L
Goma E
Morelle J
Espinosa M
Morales E
Avila A
Cabello V
Ariceta G
Chocron S
Manrique J
Barros X
Martin N
Huerta A
Fraga-Rodriguez GM
Cao M
Martin M
Romera AM
Moreso F
Manonelles A
Gratacos E
Pereira A
Campistol JM
Diaz-Ricart M
author_role author
author2 Blasco M
Molina P
Lozano M
Praga M
Torramade-Moix S
Martinez-Sanchez J
Cid J
Escolar G
Carreras E
Paules C
Crispi F
Quintana LF
Poch E
Rodas L
Goma E
Morelle J
Espinosa M
Morales E
Avila A
Cabello V
Ariceta G
Chocron S
Manrique J
Barros X
Martin N
Huerta A
Fraga-Rodriguez GM
Cao M
Martin M
Romera AM
Moreso F
Manonelles A
Gratacos E
Pereira A
Campistol JM
Diaz-Ricart M
author2_role author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
dc.subject.none.fl_str_mv humans
pregnancy
female
atypical hemolytic uremic syndrome
complement membrane
HELLP syndrome
complement C9
eculizumab
complement pathway
alternative
pre-eclampsia
hypertension
malignant
fibrin
antibodies
monoclonal
humanized
complement system proteins
complement activation
thrombotic microangiopathies
recurrence
fluorescent antibody technique
endothelial cells
topic humans
pregnancy
female
atypical hemolytic uremic syndrome
complement membrane
HELLP syndrome
complement C9
eculizumab
complement pathway
alternative
pre-eclampsia
hypertension
malignant
fibrin
antibodies
monoclonal
humanized
complement system proteins
complement activation
thrombotic microangiopathies
recurrence
fluorescent antibody technique
endothelial cells
description Background and objectivesAtypical hemolytic uremic syndrome is a form of thrombotic microangiopathy caused by dysregulation of the alternative complement pathway. There is evidence showing complement activation in other thrombotic microangiopathies. The aim of this study was to evaluate complement activation in different thrombotic microangiopathies and to monitor treatment response.Design, setting, participants, & measurementsComplement activation was assessed by exposing endothelial cells to sera or activated-patient plasma?citrated plasma mixed with a control sera pool (1:1)?to analyze C5b-9 deposits by immunofluorescence. Patients with atypical hemolytic uremic syndrome (n=34) at different stages of the disease, HELLP syndrome (a pregnancy complication characterized by hemolysis, elevated liver enzymes, and low platelet count) or severe preeclampsia (n=10), and malignant hypertension (n=5) were included.ResultsAcute phase atypical hemolytic uremic syndrome?activated plasma induced an increased C5b-9 deposition on endothelial cells. Standard and lower doses of eculizumab inhibited C5b-9 deposition in all patients with atypical hemolytic uremic syndrome, except in two who showed partial remission and clinical relapse. Significant fibrin formation was observed together with C5b-9 deposition. Results obtained using activated-plasma samples were more marked and reproducible than those obtained with sera. C5b-9 deposition was also increased with samples from patients with HELLP (all cases) and preeclampsia (90%) at disease onset. This increase was sustained in those with HELLP after 40 days, and levels normalized in patients with both HELLP and preeclampsia after 6?9 months. Complement activation in those with malignant hypertension was at control levels.ConclusionsThe proposed methodology identifies complement overactivation in patients with atypical hemolytic uremic syndrome at acute phase and in other diseases such as HELLP syndrome and preeclampsia. Moreover, it is sensitive enough to individually assess the efficiency of the C5 inhibition treatment.
publishDate 2019
dc.date.none.fl_str_mv 2019
dc.type.none.fl_str_mv info:eu-repo/semantics/article
info:eu-repo/semantics/publishedVersion
format article
status_str publishedVersion
dc.identifier.none.fl_str_mv https://fisabio.portalinvestigacion.com/publicaciones/4518
url https://fisabio.portalinvestigacion.com/publicaciones/4518
dc.language.none.fl_str_mv Inglés
language_invalid_str_mv Inglés
dc.rights.none.fl_str_mv info:eu-repo/semantics/openAccess
eu_rights_str_mv openAccess
dc.publisher.none.fl_str_mv American Society of Nephrology
publisher.none.fl_str_mv American Society of Nephrology
dc.source.none.fl_str_mv Clinical journal of the American Society of Nephrology
ISSN: 1555905X
ISSNe: 15559041
reponame:r-FISABIO. Repositorio Institucional de Producción Científica
instname:Fundación para el Fomento de la Investigación Sanitaria y Biomédica de la Comunitat Valenciana (FISABIO)
instname_str Fundación para el Fomento de la Investigación Sanitaria y Biomédica de la Comunitat Valenciana (FISABIO)
reponame_str r-FISABIO. Repositorio Institucional de Producción Científica
collection r-FISABIO. Repositorio Institucional de Producción Científica
repository.name.fl_str_mv
repository.mail.fl_str_mv
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