Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies

The clinical outcomes of primary immunodeficiencies (PIDs) are greatly improved by accurate diagnosis early in life. However, it is not common to consider PIDs before the manifestation of severe clinical symptoms. Including PIDs in the nation-wide newborn screening programs will potentially improve...

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Autores: Dezfouli, Mahya, Bergstrom, Sofia, Skattum, Lillemor, Abolhassani, Hassan, Neiman, Maja, Torabi-Rahvar, Monireh, Franco Jarava, Clara, Martin-Nalda, Andrea, Ferrer Balaguer, Juana Maria, Slade, Charlotte A., Roos, Anja, Fernandez Pereira, Luis M., Lopez-Trascasa, Margarita, Gonzalez-Granado, Luis, I, Allende-Martinez, Luis M., Mizuno, Yumi, Yoshida, Yusuke, Friman, Vanda, Lundgren, Asa, Aghamohammadi, Asghar, Rezaei, Nima, Hernandez-Guerra, Manuel, von Dobeln, Ulrika, Truedsson, Lennart, Hara, Toshiro, Nonoyama, Shigeaki, Schwenk, Jochen M., Nilsson, Peter, Hammarstrom, Lennart
Tipo de recurso: artículo
Fecha de publicación:2020
País:España
Institución:Conselleria de Salut i Consum del Govern de les Illes Balears
Repositorio:Docusalut
Idioma:inglés
OAI Identifier:oai:docusalut.com:20.500.13003/11343
Acceso en línea:https://hdl.handle.net/20.500.13003/11343
Access Level:acceso abierto
Palabra clave:Early Diagnosis
Phagocytosis
Hereditary Complement Deficiency Diseases
Immunologic Deficiency Syndromes
Infant, Newborn
Retrospective Studies
Phagocyte Bactericidal Dysfunction
Phagocytes
Humans
Neonatal Screening
Tamizaje Neonatal
Enfermedades por Deficiencia de Complemento Hereditario
Humanos
Fagocitosis
Recién Nacido
Fagocitos
Disfunción de Fagocito Bactericida
Estudios Retrospectivos
Diagnóstico Precoz
Síndromes de Inmunodeficiencia
primary immunodeficiency
complement deficiencies
phagocytic disorders
presymptomatic diagnosis
newborn screening
dried blood spot
protein profiling
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spelling Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte DeficienciesDezfouli, MahyaBergstrom, SofiaSkattum, LillemorAbolhassani, HassanNeiman, MajaTorabi-Rahvar, MonirehFranco Jarava, ClaraMartin-Nalda, AndreaFerrer Balaguer, Juana MariaSlade, Charlotte A.Roos, AnjaFernandez Pereira, Luis M.Lopez-Trascasa, MargaritaGonzalez-Granado, Luis, IAllende-Martinez, Luis M.Mizuno, YumiYoshida, YusukeFriman, VandaLundgren, AsaAghamohammadi, AsgharRezaei, NimaHernandez-Guerra, Manuelvon Dobeln, UlrikaTruedsson, LennartHara, ToshiroNonoyama, ShigeakiSchwenk, Jochen M.Nilsson, PeterHammarstrom, LennartEarly DiagnosisPhagocytosisHereditary Complement Deficiency DiseasesImmunologic Deficiency SyndromesInfant, NewbornRetrospective StudiesPhagocyte Bactericidal DysfunctionPhagocytesHumansNeonatal ScreeningTamizaje NeonatalEnfermedades por Deficiencia de Complemento HereditarioHumanosFagocitosisRecién NacidoFagocitosDisfunción de Fagocito BactericidaEstudios RetrospectivosDiagnóstico PrecozSíndromes de Inmunodeficienciaprimary immunodeficiencycomplement deficienciesphagocytic disorderspresymptomatic diagnosisnewborn screeningdried blood spotprotein profilingThe clinical outcomes of primary immunodeficiencies (PIDs) are greatly improved by accurate diagnosis early in life. However, it is not common to consider PIDs before the manifestation of severe clinical symptoms. Including PIDs in the nation-wide newborn screening programs will potentially improve survival and provide better disease management and preventive care in PID patients. This calls for the detection of disease biomarkers in blood and the use of dried blood spot samples, which is a part of routine newborn screening programs worldwide. Here, we developed a newborn screening method based on multiplex protein profiling for parallel diagnosis of 22 innate immunodeficiencies affecting the complement system and respiratory burst function in phagocytosis. The proposed method uses a small fraction of eluted blood from dried blood spots and is applicable for population-scale performance. The diagnosis method is validated through a retrospective screening of immunodeficient patient samples. This diagnostic approach can pave the way for an earlier, more comprehensive and accurate diagnosis of complement and phagocytic disorders, which ultimately lead to a healthy and active life for the PID patients.Frontiers Media Sa20202020-03-1720202020-03-17research articlehttp://purl.org/coar/resource_type/c_2df8fbb1info:eu-repo/semantics/articleapplication/pdfhttps://hdl.handle.net/20.500.13003/11343reponame:Docusalutinstname:Conselleria de Salut i Consum del Govern de les Illes BalearsInglésengopen accesshttp://purl.org/coar/access_right/c_abf2Attribution 4.0 Internationalhttp://creativecommons.org/licenses/by/4.0/info:eu-repo/semantics/openAccessoai:docusalut.com:20.500.13003/113432026-06-22T12:44:07Z
dc.title.none.fl_str_mv Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
title Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
spellingShingle Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
Dezfouli, Mahya
Early Diagnosis
Phagocytosis
Hereditary Complement Deficiency Diseases
Immunologic Deficiency Syndromes
Infant, Newborn
Retrospective Studies
Phagocyte Bactericidal Dysfunction
Phagocytes
Humans
Neonatal Screening
Tamizaje Neonatal
Enfermedades por Deficiencia de Complemento Hereditario
Humanos
Fagocitosis
Recién Nacido
Fagocitos
Disfunción de Fagocito Bactericida
Estudios Retrospectivos
Diagnóstico Precoz
Síndromes de Inmunodeficiencia
primary immunodeficiency
complement deficiencies
phagocytic disorders
presymptomatic diagnosis
newborn screening
dried blood spot
protein profiling
title_short Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
title_full Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
title_fullStr Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
title_full_unstemmed Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
title_sort Newborn Screening for Presymptomatic Diagnosis of Complement and Phagocyte Deficiencies
dc.creator.none.fl_str_mv Dezfouli, Mahya
Bergstrom, Sofia
Skattum, Lillemor
Abolhassani, Hassan
Neiman, Maja
Torabi-Rahvar, Monireh
Franco Jarava, Clara
Martin-Nalda, Andrea
Ferrer Balaguer, Juana Maria
Slade, Charlotte A.
Roos, Anja
Fernandez Pereira, Luis M.
Lopez-Trascasa, Margarita
Gonzalez-Granado, Luis, I
Allende-Martinez, Luis M.
Mizuno, Yumi
Yoshida, Yusuke
Friman, Vanda
Lundgren, Asa
Aghamohammadi, Asghar
Rezaei, Nima
Hernandez-Guerra, Manuel
von Dobeln, Ulrika
Truedsson, Lennart
Hara, Toshiro
Nonoyama, Shigeaki
Schwenk, Jochen M.
Nilsson, Peter
Hammarstrom, Lennart
author Dezfouli, Mahya
author_facet Dezfouli, Mahya
Bergstrom, Sofia
Skattum, Lillemor
Abolhassani, Hassan
Neiman, Maja
Torabi-Rahvar, Monireh
Franco Jarava, Clara
Martin-Nalda, Andrea
Ferrer Balaguer, Juana Maria
Slade, Charlotte A.
Roos, Anja
Fernandez Pereira, Luis M.
Lopez-Trascasa, Margarita
Gonzalez-Granado, Luis, I
Allende-Martinez, Luis M.
Mizuno, Yumi
Yoshida, Yusuke
Friman, Vanda
Lundgren, Asa
Aghamohammadi, Asghar
Rezaei, Nima
Hernandez-Guerra, Manuel
von Dobeln, Ulrika
Truedsson, Lennart
Hara, Toshiro
Nonoyama, Shigeaki
Schwenk, Jochen M.
Nilsson, Peter
Hammarstrom, Lennart
author_role author
author2 Bergstrom, Sofia
Skattum, Lillemor
Abolhassani, Hassan
Neiman, Maja
Torabi-Rahvar, Monireh
Franco Jarava, Clara
Martin-Nalda, Andrea
Ferrer Balaguer, Juana Maria
Slade, Charlotte A.
Roos, Anja
Fernandez Pereira, Luis M.
Lopez-Trascasa, Margarita
Gonzalez-Granado, Luis, I
Allende-Martinez, Luis M.
Mizuno, Yumi
Yoshida, Yusuke
Friman, Vanda
Lundgren, Asa
Aghamohammadi, Asghar
Rezaei, Nima
Hernandez-Guerra, Manuel
von Dobeln, Ulrika
Truedsson, Lennart
Hara, Toshiro
Nonoyama, Shigeaki
Schwenk, Jochen M.
Nilsson, Peter
Hammarstrom, Lennart
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
dc.contributor.none.fl_str_mv
dc.subject.none.fl_str_mv Early Diagnosis
Phagocytosis
Hereditary Complement Deficiency Diseases
Immunologic Deficiency Syndromes
Infant, Newborn
Retrospective Studies
Phagocyte Bactericidal Dysfunction
Phagocytes
Humans
Neonatal Screening
Tamizaje Neonatal
Enfermedades por Deficiencia de Complemento Hereditario
Humanos
Fagocitosis
Recién Nacido
Fagocitos
Disfunción de Fagocito Bactericida
Estudios Retrospectivos
Diagnóstico Precoz
Síndromes de Inmunodeficiencia
primary immunodeficiency
complement deficiencies
phagocytic disorders
presymptomatic diagnosis
newborn screening
dried blood spot
protein profiling
topic Early Diagnosis
Phagocytosis
Hereditary Complement Deficiency Diseases
Immunologic Deficiency Syndromes
Infant, Newborn
Retrospective Studies
Phagocyte Bactericidal Dysfunction
Phagocytes
Humans
Neonatal Screening
Tamizaje Neonatal
Enfermedades por Deficiencia de Complemento Hereditario
Humanos
Fagocitosis
Recién Nacido
Fagocitos
Disfunción de Fagocito Bactericida
Estudios Retrospectivos
Diagnóstico Precoz
Síndromes de Inmunodeficiencia
primary immunodeficiency
complement deficiencies
phagocytic disorders
presymptomatic diagnosis
newborn screening
dried blood spot
protein profiling
description The clinical outcomes of primary immunodeficiencies (PIDs) are greatly improved by accurate diagnosis early in life. However, it is not common to consider PIDs before the manifestation of severe clinical symptoms. Including PIDs in the nation-wide newborn screening programs will potentially improve survival and provide better disease management and preventive care in PID patients. This calls for the detection of disease biomarkers in blood and the use of dried blood spot samples, which is a part of routine newborn screening programs worldwide. Here, we developed a newborn screening method based on multiplex protein profiling for parallel diagnosis of 22 innate immunodeficiencies affecting the complement system and respiratory burst function in phagocytosis. The proposed method uses a small fraction of eluted blood from dried blood spots and is applicable for population-scale performance. The diagnosis method is validated through a retrospective screening of immunodeficient patient samples. This diagnostic approach can pave the way for an earlier, more comprehensive and accurate diagnosis of complement and phagocytic disorders, which ultimately lead to a healthy and active life for the PID patients.
publishDate 2020
dc.date.none.fl_str_mv 2020
2020-03-17
2020
2020-03-17
dc.type.none.fl_str_mv research article
http://purl.org/coar/resource_type/c_2df8fbb1
dc.type.openaire.fl_str_mv info:eu-repo/semantics/article
format article
dc.identifier.none.fl_str_mv https://hdl.handle.net/20.500.13003/11343
url https://hdl.handle.net/20.500.13003/11343
dc.language.none.fl_str_mv Inglés
eng
language_invalid_str_mv Inglés
language eng
dc.rights.none.fl_str_mv open access
http://purl.org/coar/access_right/c_abf2
Attribution 4.0 International
http://creativecommons.org/licenses/by/4.0/
dc.rights.openaire.fl_str_mv info:eu-repo/semantics/openAccess
rights_invalid_str_mv open access
http://purl.org/coar/access_right/c_abf2
Attribution 4.0 International
http://creativecommons.org/licenses/by/4.0/
eu_rights_str_mv openAccess
dc.format.none.fl_str_mv application/pdf
dc.publisher.none.fl_str_mv Frontiers Media Sa
publisher.none.fl_str_mv Frontiers Media Sa
dc.source.none.fl_str_mv reponame:Docusalut
instname:Conselleria de Salut i Consum del Govern de les Illes Balears
instname_str Conselleria de Salut i Consum del Govern de les Illes Balears
reponame_str Docusalut
collection Docusalut
repository.name.fl_str_mv
repository.mail.fl_str_mv
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