Literature DB >> 8471764

Immune reconstitution in severe combined immunodeficiency disease after lectin-treated, T-cell-depleted haplocompatible bone marrow transplantation.

Y Dror1, R Gallagher, D W Wara, B W Colombe, A Merino, M Benkerrou, M J Cowan.   

Abstract

We describe our 9-year experience with lectin-treated T-cell-depleted haplocompatible parental bone marrow transplantation (BMT) for 24 patients with severe combined immunodeficiency disease (SCID). Nineteen of 21 evaluable patients had T-cell engraftment; 2 of 11 patients tested had B-cell and monocyte engraftment. Fourteen of 24 (58%) patients are alive 7 months to 9.8 years post-BMT. Seventeen of 24 patients received pretransplant conditioning with chemotherapy and/or total body irradiation, and 8 of 24 received more than one transplant. Patients who received conditioning had a survival rate of 61% versus 57% for those who received no conditioning. None received graft-versus-host disease (GVHD) prophylaxis and no patient had acute or chronic GVHD greater than grade I. Kinetics and follow-up of immune recovery were analyzed in 14 patients who are greater than 1 year from transplant. Half of the patients showed evidence of T-cell function by 3 months and normal T-cell function by 4 to 7 months post-BMT. On average, T-cell numbers and subsets became normal 10 to 12 months posttransplant. Recovery of B-cell function was more delayed, although in most patients B-cell numbers and IgM levels were normal by 12 months post-BMT. B-cell function, as determined by isohemagglutinin titers or specific antibodies to pneumococcal polysaccharide, keyhole limpet hemocyanin, or tetanus toxoid, became normal in 10 of 14 patients 2 to 8 years post-BMT. Seven of the 14 are off gammaglobulin therapy. Production of isohemagglutinins tended to predict recovery of antibody response to pneumococcal polysaccharide (P < .064). Based on these results, we believe that haplocompatible BMT is an effective, curative treatment for patients with SCID who lack an HLA-matched related donor.

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Year:  1993        PMID: 8471764

Source DB:  PubMed          Journal:  Blood        ISSN: 0006-4971            Impact factor:   22.113


  24 in total

1.  Maternal mosaicism for a novel interleukin-2 receptor gamma-chain mutation causing X-linked severe combined immunodeficiency in a Navajo kindred.

Authors:  A S O'Marcaigh; J M Puck; A E Pepper; K De Santes; M J Cowan
Journal:  J Clin Immunol       Date:  1997-01       Impact factor: 8.317

Review 2.  Stem cell transplantation for immunodeficiency.

Authors:  A Fischer; E Haddad; N Jabado; J L Casanova; S Blanche; F Le Deist; M Cavazzana-Calvo
Journal:  Springer Semin Immunopathol       Date:  1998

3.  Genotype, phenotype, and outcomes of nine patients with T-B+NK+ SCID.

Authors:  Grace P Yu; Kari C Nadeau; David R Berk; Geneviève de Saint Basile; Nathalie Lambert; Perrine Knapnougel; Joseph Roberts; Kristina Kavanau; Elizabeth Dunn; E Richard Stiehm; David B Lewis; Dale T Umetsu; Jennifer M Puck; Morton J Cowan
Journal:  Pediatr Transplant       Date:  2011-08-23

4.  The gene for severe combined immunodeficiency disease in Athabascan-speaking Native Americans is located on chromosome 10p.

Authors:  L Li; D Drayna; D Hu; A Hayward; S Gahagan; H Pabst; M J Cowan
Journal:  Am J Hum Genet       Date:  1998-01       Impact factor: 11.025

Review 5.  Current Knowledge and Priorities for Future Research in Late Effects after Hematopoietic Stem Cell Transplantation (HCT) for Severe Combined Immunodeficiency Patients: A Consensus Statement from the Second Pediatric Blood and Marrow Transplant Consortium International Conference on Late Effects after Pediatric HCT.

Authors:  Jennifer Heimall; Jennifer Puck; Rebecca Buckley; Thomas A Fleisher; Andrew R Gennery; Benedicte Neven; Mary Slatter; Elie Haddad; Luigi D Notarangelo; K Scott Baker; Andrew C Dietz; Christine Duncan; Michael A Pulsipher; Mort J Cowan
Journal:  Biol Blood Marrow Transplant       Date:  2017-01-06       Impact factor: 5.742

6.  Hematopoietic Stem Cell Transplantation for Severe Combined Immunodeficiency.

Authors:  Justin T Wahlstrom; Christopher C Dvorak; Morton J Cowan
Journal:  Curr Pediatr Rep       Date:  2015-03-01

7.  Allogeneic hematopoietic cell transplantation for primary immune deficiency diseases: current status and critical needs.

Authors:  Linda M Griffith; Morton J Cowan; Donald B Kohn; Luigi D Notarangelo; Jennifer M Puck; Kirk R Schultz; Rebecca H Buckley; Mary Eapen; Naynesh R Kamani; Richard J O'Reilly; Robertson Parkman; Chaim M Roifman; Kathleen E Sullivan; Alexandra H Filipovich; Thomas A Fleisher; William T Shearer
Journal:  J Allergy Clin Immunol       Date:  2008-11-06       Impact factor: 10.793

8.  Anti-tetanus toxoid antibody production after mismatched T cell-depleted bone marrow transplantation.

Authors:  M Benkerrou; D W Wara; M Elder; Y Dror; A Merino; B W Colombe; M Garovoy; M J Cowan
Journal:  J Clin Immunol       Date:  1994-03       Impact factor: 8.317

9.  Transplacental maternal engraftment and posttransplantation graft-versus-host disease in children with severe combined immunodeficiency.

Authors:  Justin Wahlstrom; Kiran Patel; Erik Eckhert; Denice Kong; Biljana Horn; Morton J Cowan; Christopher C Dvorak
Journal:  J Allergy Clin Immunol       Date:  2016-06-16       Impact factor: 10.793

10.  Establishing diagnostic criteria for severe combined immunodeficiency disease (SCID), leaky SCID, and Omenn syndrome: the Primary Immune Deficiency Treatment Consortium experience.

Authors:  William T Shearer; Elizabeth Dunn; Luigi D Notarangelo; Christopher C Dvorak; Jennifer M Puck; Brent R Logan; Linda M Griffith; Donald B Kohn; Richard J O'Reilly; Thomas A Fleisher; Sung-Yun Pai; Caridad A Martinez; Rebecca H Buckley; Morton J Cowan
Journal:  J Allergy Clin Immunol       Date:  2013-11-28       Impact factor: 10.793

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