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Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient

We describe the case of a child affected by severe combined immunodeficiency (SCID) with adenosine deaminase (ADA) deficiency showing a maternal T-cell engraftment, a finding that has never been reported before. The presence of engrafted maternal T cells was misleading. Although ADA enzymatic levels...

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Autores principales: Lanfranchi, Arnalda, Lougaris, Vassilios, Notarangelo, Lucia Dora, Soncini, Elena, Comini, Marta, Beghin, Alessandra, Bolda, Federica, Montanelli, Alessandro, Imberti, Luisa, Porta, Fulvio
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier Inc. 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7106042/
https://www.ncbi.nlm.nih.gov/pubmed/29355610
http://dx.doi.org/10.1016/j.clim.2018.01.004
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author Lanfranchi, Arnalda
Lougaris, Vassilios
Notarangelo, Lucia Dora
Soncini, Elena
Comini, Marta
Beghin, Alessandra
Bolda, Federica
Montanelli, Alessandro
Imberti, Luisa
Porta, Fulvio
author_facet Lanfranchi, Arnalda
Lougaris, Vassilios
Notarangelo, Lucia Dora
Soncini, Elena
Comini, Marta
Beghin, Alessandra
Bolda, Federica
Montanelli, Alessandro
Imberti, Luisa
Porta, Fulvio
author_sort Lanfranchi, Arnalda
collection PubMed
description We describe the case of a child affected by severe combined immunodeficiency (SCID) with adenosine deaminase (ADA) deficiency showing a maternal T-cell engraftment, a finding that has never been reported before. The presence of engrafted maternal T cells was misleading. Although ADA enzymatic levels were suggestive of ADA-SCID, the child did not present the classical signs of ADA deficiency; therefore, the initial diagnosis was of a conventional SCID. However, ADA toxic metabolites and molecular characterization confirmed this diagnosis. Polyethylene glycol-modified bovine (PEG) ADA therapy progressively decreased the number of maternal engrafted T cells. The child was grafted with full bone marrow from a matched unrelated donor, after a reduced conditioning regimen, and the result was the complete immunological reconstitution.
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spelling pubmed-71060422020-03-31 Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient Lanfranchi, Arnalda Lougaris, Vassilios Notarangelo, Lucia Dora Soncini, Elena Comini, Marta Beghin, Alessandra Bolda, Federica Montanelli, Alessandro Imberti, Luisa Porta, Fulvio Clin Immunol Article We describe the case of a child affected by severe combined immunodeficiency (SCID) with adenosine deaminase (ADA) deficiency showing a maternal T-cell engraftment, a finding that has never been reported before. The presence of engrafted maternal T cells was misleading. Although ADA enzymatic levels were suggestive of ADA-SCID, the child did not present the classical signs of ADA deficiency; therefore, the initial diagnosis was of a conventional SCID. However, ADA toxic metabolites and molecular characterization confirmed this diagnosis. Polyethylene glycol-modified bovine (PEG) ADA therapy progressively decreased the number of maternal engrafted T cells. The child was grafted with full bone marrow from a matched unrelated donor, after a reduced conditioning regimen, and the result was the complete immunological reconstitution. Elsevier Inc. 2018-08 2018-05-07 /pmc/articles/PMC7106042/ /pubmed/29355610 http://dx.doi.org/10.1016/j.clim.2018.01.004 Text en © 2018 Elsevier Inc. All rights reserved. Since January 2020 Elsevier has created a COVID-19 resource centre with free information in English and Mandarin on the novel coronavirus COVID-19. The COVID-19 resource centre is hosted on Elsevier Connect, the company's public news and information website. Elsevier hereby grants permission to make all its COVID-19-related research that is available on the COVID-19 resource centre - including this research content - immediately available in PubMed Central and other publicly funded repositories, such as the WHO COVID database with rights for unrestricted research re-use and analyses in any form or by any means with acknowledgement of the original source. These permissions are granted for free by Elsevier for as long as the COVID-19 resource centre remains active.
spellingShingle Article
Lanfranchi, Arnalda
Lougaris, Vassilios
Notarangelo, Lucia Dora
Soncini, Elena
Comini, Marta
Beghin, Alessandra
Bolda, Federica
Montanelli, Alessandro
Imberti, Luisa
Porta, Fulvio
Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient
title Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient
title_full Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient
title_fullStr Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient
title_full_unstemmed Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient
title_short Maternal T-cell engraftment impedes with diagnosis of a SCID-ADA patient
title_sort maternal t-cell engraftment impedes with diagnosis of a scid-ada patient
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7106042/
https://www.ncbi.nlm.nih.gov/pubmed/29355610
http://dx.doi.org/10.1016/j.clim.2018.01.004
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