Literature DB >> 31222511

Immune reconstitution after HSCT in SCID-a cohort of conditioned and unconditioned patients.

Uri Manor1, Atar Lev2, Amos J Simon2, Daphna Hutt3, Amos Toren1,3, Bella Bielorai1,3, Lior Goldberg1,2, Tali Stauber1,2, Raz Somech4,5,6.   

Abstract

Allogeneic hematopoietic stem cell transplantation (HSCT) is the effective mean of immune restoration in severe combined immunodefiency (SCID). Usually, HSCT without cytoreductive conditioning is attempted. Nevertheless, conditioning procedures are still preferred in a subset of patients. Herein, we describe the immunological outcome in a cohort of conditioned and unconditioned patients, from diagnosis, through transplantation, to follow-up. This retrospective study was conducted on 17 patients with SCID (10 conditioned, 7 unconditioned) who later underwent HSCT. Immune reconstitution was assessed in the post-transplant year by quantification of T cell receptor excision circles (TRECs) and kappa-deleting recombination excision circles (KRECs), among additional laboratory and clinical evaluations. Unconditioned patients were diagnosed and transplanted earlier. TREC and KREC quantification showed a gradual increase in both groups, with higher levels in the conditioned group. Engraftment percentages differed drastically between groups, favoring the conditioned group. Unconditioned patients were significantly more dependent on intravenous immunoglobulins (IVIGs). One patient from each group succumbed to disease complications. Conditioning demonstrated superior laboratorial outcomes. Patients with unique characteristics (i.e., consanguinity, Bacillus Calmette-Guérin vaccination, impaired access to IVIG) may require personalized considerations. The effort to implement secondary prevention of SCID with newborn screening should continue.

Entities:  

Keywords:  Conditioning; Hematopoietic stem cell transplantation (HSCT); Kappa-deleting recombination excision circles (KRECs); Primary immunodeficiency (PID); Severe combined immunodeficiency (SCID); T cell receptor excision circles (TRECs)

Mesh:

Substances:

Year:  2019        PMID: 31222511     DOI: 10.1007/s12026-019-09081-z

Source DB:  PubMed          Journal:  Immunol Res        ISSN: 0257-277X            Impact factor:   2.829


  29 in total

1.  How I treat severe combined immunodeficiency.

Authors:  H Bobby Gaspar; Waseem Qasim; E Graham Davies; Kanchan Rao; Persis J Amrolia; Paul Veys
Journal:  Blood       Date:  2013-10-10       Impact factor: 22.113

2.  A comparison of fluorescent in situ hybridization and multiplex short tandem repeat polymerase chain reaction for quantifying chimerism after stem cell transplantation.

Authors:  Ismael Buño; Paola Nava; Ainhoa Simón; Milagros González-Rivera; José Luis Jiménez; Pascual Balsalobre; David Serrano; Rafael Carrión; Alfonso Gómez-Pineda; José Luis Díez-Martín
Journal:  Haematologica       Date:  2005-10       Impact factor: 9.941

Review 3.  Systematic evidence review of newborn screening and treatment of severe combined immunodeficiency.

Authors:  Ellen A Lipstein; Sienna Vorono; Marsha F Browning; Nancy S Green; Alex R Kemper; Alixandra A Knapp; Lisa A Prosser; James M Perrin
Journal:  Pediatrics       Date:  2010-04-19       Impact factor: 7.124

Review 4.  Bone marrow transplantation for primary immunodeficiency diseases.

Authors:  Paul Szabolcs; Marina Cavazzana-Calvo; Alain Fischer; Paul Veys
Journal:  Pediatr Clin North Am       Date:  2010-02       Impact factor: 3.278

Review 5.  B-cell reconstitution for SCID: should a conditioning regimen be used in SCID treatment?

Authors:  Elie Haddad; Sandrine Leroy; Rebecca H Buckley
Journal:  J Allergy Clin Immunol       Date:  2013-03-05       Impact factor: 10.793

Review 6.  Use of V(D)J recombination excision circles to identify T- and B-cell defects and to monitor the treatment in primary and acquired immunodeficiencies.

Authors:  Federico Serana; Marco Chiarini; Cinzia Zanotti; Alessandra Sottini; Diego Bertoli; Andrea Bosio; Luigi Caimi; Luisa Imberti
Journal:  J Transl Med       Date:  2013-05-09       Impact factor: 5.531

7.  First Year of Israeli Newborn Screening for Severe Combined Immunodeficiency-Clinical Achievements and Insights.

Authors:  Erez Rechavi; Atar Lev; Amos J Simon; Tali Stauber; Suha Daas; Talia Saraf-Levy; Arnon Broides; Amit Nahum; Nufar Marcus; Suhair Hanna; Polina Stepensky; Ori Toker; Ilan Dalal; Amos Etzioni; Shlomo Almashanu; Raz Somech
Journal:  Front Immunol       Date:  2017-11-06       Impact factor: 7.561

8.  Bone marrow transplantation for severe combined immune deficiency.

Authors:  Eyal Grunebaum; Evelina Mazzolari; Fulvio Porta; Daniela Dallera; Adelle Atkinson; Brenda Reid; Luigi D Notarangelo; Chaim M Roifman
Journal:  JAMA       Date:  2006-02-01       Impact factor: 56.272

9.  PID comes full circle: applications of V(D)J recombination excision circles in research, diagnostics and newborn screening of primary immunodeficiency disorders.

Authors:  Menno C van Zelm; Mirjam van der Burg; Anton W Langerak; Jacques J M van Dongen
Journal:  Front Immunol       Date:  2011-05-04       Impact factor: 7.561

10.  Utilization of TREC and KREC quantification for the monitoring of early T- and B-cell neogenesis in adult patients after allogeneic hematopoietic stem cell transplantation.

Authors:  Angela Mensen; Christoph Ochs; Andrea Stroux; Friedrich Wittenbecher; Martin Szyska; Luisa Imberti; Simon Fillatreau; Lutz Uharek; Renate Arnold; Bernd Dörken; Andreas Thiel; Carmen Scheibenbogen; Il-Kang Na
Journal:  J Transl Med       Date:  2013-08-14       Impact factor: 5.531

View more
  2 in total

1.  Long-Term Immune Recovery After Hematopoietic Stem Cell Transplantation for ADA Deficiency: a Single-Center Experience.

Authors:  H Bobby Gaspar; Claire Booth; Alexandra Y Kreins; Helena F Velasco; Kai-Ning Cheong; Kanchan Rao; Paul Veys; Austen Worth
Journal:  J Clin Immunol       Date:  2021-10-16       Impact factor: 8.317

Review 2.  Considerations in Preparative Regimen Selection to Minimize Rejection in Pediatric Hematopoietic Transplantation in Non-Malignant Diseases.

Authors:  Robert J Hayashi
Journal:  Front Immunol       Date:  2020-10-19       Impact factor: 7.561

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.