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

Authors :
Alonso, Laura
Méndez-Echevarría, Ana
Rudilla, Francesc
Mozo, Yasmina
Soler-Palacin, Pere
Sisinni, Luisa
Bueno, David
Riviere, Jacques
de Paz, Raquel
Sánchez-Zapardiel, Elena
Querol, Sergi
Rodriguez-Pena, Rebeca
López-Granados, Eduardo
Gimeno, Ramón
Díaz de Heredia, Cristina
Pérez-Martínez, Antonio
Source :
Journal of Clinical Immunology. May2021, Vol. 41 Issue 4, p748-755. 8p.
Publication Year :
2021

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. [ABSTRACT FROM AUTHOR]

Details

Language :
English
ISSN :
02719142
Volume :
41
Issue :
4
Database :
Academic Search Index
Journal :
Journal of Clinical Immunology
Publication Type :
Academic Journal
Accession number :
149989617
Full Text :
https://doi.org/10.1007/s10875-020-00961-w