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Erschienen in: Journal of Clinical Immunology 4/2021

18.01.2021 | Original Article

Failure of Viral-Specific T Cells Administered in Pre-transplant Settings in Children with Inborn Errors of Immunity

verfasst von: Laura Alonso, Ana Méndez-Echevarría, Francesc Rudilla, Yasmina Mozo, Pere Soler-Palacin, Luisa Sisinni, David Bueno, Jacques Riviere, Raquel de Paz, Elena Sánchez-Zapardiel, Sergi Querol, Rebeca Rodriguez-Pena, Eduardo López-Granados, Ramón Gimeno, Cristina Díaz de Heredia, Antonio Pérez-Martínez

Erschienen in: Journal of Clinical Immunology | Ausgabe 4/2021

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Abstract

Purpose

Use of adoptive immunotherapy with virus-specific T cells (VST) in patients with inborn errors of immunity prior to hematopoietic stem cell transplantation (HSCT) has been reported in few patients. We report our experience, reviewing all the cases previously reported.

Methods

We report four children with inborn errors of immunity who received VST infusion in a pre-HSCT setting in two reference centers in Spain and review all inborn errors of immunity cases previously reported.

Results

Taking into account our four cases, nine children have been reported to receive VST prior to HSCT to date: 3 severe combined immunodeficiency, 2 CTPS1 deficiency, 1 dyskeratosis congenital, 1 ORAI1 deficiency, 1 Rothmund-Thomson syndrome, and 1 combined immunodeficiency without confirmed genetic defect. In four patients, immunotherapy resulted in clinical improvement, allowing to proceed to HSCT. In these cases, the infusion was started closely to viral diagnosis [mean time 28 days (IQR; 17–52 days)], and the VST was followed shortly thereafter by HSCT [mean time 28 days (IQR; 10–99 days)]. Viremia was controlled after HSCT in two cases (performed 7 and 36 days after the infusion). Multiple infusions were required in many cases. Five out of nine patients died before receiving HSCT. These patients presented with a prolonged and uncontrolled infection before VST administration [mean time from viral diagnosis to VST infusion was 176 days (IQR; 54–1687)].

Conclusions

In patients with inborn errors of immunity, the efficacy of VST for treating disseminated viral infections in pre-transplant settings seems to have a limited efficacy. However, this therapy could be used in a pre-emptive setting before severe viral disease occurs or closely to HSCT.
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Metadaten
Titel
Failure of Viral-Specific T Cells Administered in Pre-transplant Settings in Children with Inborn Errors of Immunity
verfasst von
Laura Alonso
Ana Méndez-Echevarría
Francesc Rudilla
Yasmina Mozo
Pere Soler-Palacin
Luisa Sisinni
David Bueno
Jacques Riviere
Raquel de Paz
Elena Sánchez-Zapardiel
Sergi Querol
Rebeca Rodriguez-Pena
Eduardo López-Granados
Ramón Gimeno
Cristina Díaz de Heredia
Antonio Pérez-Martínez
Publikationsdatum
18.01.2021
Verlag
Springer US
Erschienen in
Journal of Clinical Immunology / Ausgabe 4/2021
Print ISSN: 0271-9142
Elektronische ISSN: 1573-2592
DOI
https://doi.org/10.1007/s10875-020-00961-w

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