Literature DB >> 18557726

Factors affecting success of thymus transplantation for complete DiGeorge anomaly.

M L Markert1, B H Devlin, I K Chinn, E A McCarthy, Y J Li.   

Abstract

Thymus transplantation shows promise for the treatment of athymia in complete DiGeorge anomaly. This report reviews the effects of dose of thymus tissue, ABO compatibility, HLA matching, culture conditions, age of donor and immunosuppression of recipient on immune outcomes at 1 year after transplantation. Forty-nine athymic subjects have been treated with cultured postnatal allogeneic thymus tissue; 36 (73%) survive with only one subject on immunosuppression at 1.5 years. Of 31 surviving subjects more than 1 year after transplantation, 30 (97%) developed naive T cells, T-cell proliferative responses to mitogens and a diverse T-cell receptor beta variable (TCRBV) repertoire. The dose of thymus tissue, HLA matching and use of immunosuppression had nonsignificant effects on these outcome variables. Removal of deoxyguanosine from culture medium and length of culture did not adversely affect outcomes. Use of thymus tissue from donors over 1 month of age, versus under 1 month, resulted in higher total T-cell numbers (p = 0.03). However, this finding must be confirmed in a prospective trial. Although subtle immune effects may yet be associated with some of the factors tested, it is remarkable that consistently good immune outcomes result despite variation in dose, HLA matching and use of immunosuppression.

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Year:  2008        PMID: 18557726      PMCID: PMC3667673          DOI: 10.1111/j.1600-6143.2008.02301.x

Source DB:  PubMed          Journal:  Am J Transplant        ISSN: 1600-6135            Impact factor:   8.086


  22 in total

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Journal:  Transplantation       Date:  1996-02-15       Impact factor: 4.939

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Authors:  P Hugo; J W Kappler; J E McCormack; P Marrack
Journal:  Proc Natl Acad Sci U S A       Date:  1993-11-01       Impact factor: 11.205

6.  Lack of correlation between impaired T cell production, immunodeficiency, and other phenotypic features in chromosome 22q11.2 deletion syndromes.

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Journal:  Clin Immunol Immunopathol       Date:  1998-02

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Journal:  Nature       Date:  1993-08-12       Impact factor: 49.962

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Journal:  J Immunol       Date:  1993-02-01       Impact factor: 5.422

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  18 in total

1.  Multicenter survey on the outcome of transplantation of hematopoietic cells in patients with the complete form of DiGeorge anomaly.

Authors:  Ales Janda; Petr Sedlacek; Manfred Hönig; Wilhelm Friedrich; Martin Champagne; Tadashi Matsumoto; Alain Fischer; Benedicte Neven; Audrey Contet; Danielle Bensoussan; Pierre Bordigoni; David Loeb; William Savage; Nada Jabado; Francisco A Bonilla; Mary A Slatter; E Graham Davies; Andrew R Gennery
Journal:  Blood       Date:  2010-06-07       Impact factor: 22.113

2.  First use of thymus transplantation therapy for FOXN1 deficiency (nude/SCID): a report of 2 cases.

Authors:  M Louise Markert; José G Marques; Bénédicte Neven; Blythe H Devlin; Elizabeth A McCarthy; Ivan K Chinn; Adriana S Albuquerque; Susana L Silva; Claudio Pignata; Geneviève de Saint Basile; Rui M Victorino; Capucine Picard; Marianne Debre; Nizar Mahlaoui; Alain Fischer; Ana E Sousa
Journal:  Blood       Date:  2010-10-26       Impact factor: 22.113

3.  Thymic microenvironment reconstitution after postnatal human thymus transplantation.

Authors:  Bin Li; Jie Li; Blythe H Devlin; M Louise Markert
Journal:  Clin Immunol       Date:  2011-04-16       Impact factor: 3.969

Review 4.  Immunological aspects of 22q11.2 deletion syndrome.

Authors:  A R Gennery
Journal:  Cell Mol Life Sci       Date:  2011-10-09       Impact factor: 9.261

Review 5.  Thymus transplantation.

Authors:  M Louise Markert; Blythe H Devlin; Elizabeth A McCarthy
Journal:  Clin Immunol       Date:  2010-03-16       Impact factor: 3.969

6.  Thymus transplantation in complete DiGeorge anomaly.

Authors:  M Louise Markert; Blythe H Devlin; Ivan K Chinn; Elizabeth A McCarthy
Journal:  Immunol Res       Date:  2009       Impact factor: 2.829

Review 7.  The immune system in infants: Relevance to xenotransplantation.

Authors:  Mohamed Bikhet; Mahmoud Morsi; Hidetaka Hara; Leslie A Rhodes; Waldemar F Carlo; David Cleveland; David K C Cooper; Hayato Iwase
Journal:  Pediatr Transplant       Date:  2020-08-26

8.  Human FOXN1-deficiency is associated with αβ double-negative and FoxP3+ T-cell expansions that are distinctly modulated upon thymic transplantation.

Authors:  Adriana S Albuquerque; José G Marques; Susana L Silva; Dario Ligeiro; Blythe H Devlin; Jacques Dutrieux; Rémi Cheynier; Claudio Pignata; Rui M M Victorino; M Louise Markert; Ana E Sousa
Journal:  PLoS One       Date:  2012-05-10       Impact factor: 3.240

9.  The dynamics of T-cell receptor repertoire diversity following thymus transplantation for DiGeorge anomaly.

Authors:  Stanca M Ciupe; Blythe H Devlin; M Louise Markert; Thomas B Kepler
Journal:  PLoS Comput Biol       Date:  2009-06-12       Impact factor: 4.475

10.  Experience with cultured thymus tissue in 105 children.

Authors:  M Louise Markert; Stephanie E Gupton; Elizabeth A McCarthy
Journal:  J Allergy Clin Immunol       Date:  2021-08-04       Impact factor: 14.290

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