Literature DB >> 3298595

Bone marrow transplantation in DiGeorge syndrome.

A B Goldsobel, A Haas, E R Stiehm.   

Abstract

A Hispanic infant girl with DiGeorge syndrome underwent successful bone marrow transplantation (BMT) at age 28 1/2 weeks. She had typical facies, a cardiac defect, hypoparathyroidism, severe T and B cell immunodeficiency, and low levels of facteur thymique serique (FTS). In vitro incubation of the peripheral blood lymphocytes with thymosin alpha 1 showed no increase in the number of T cells on two occasions. A fetal thymus for transplantation was not available, and further review of past experience with thymic cells or factors revealed inconsistent and incomplete responses. Because of the patient's worsening clinical and immunologic status, BMT was performed, with her histocompatible brother as donor. The patient has had a good clinical and immunologic response to BMT, with evidence of T cell engraftment, improved B cell function, and increased levels of serum FTS. This experience indicates that minimal thymic influence is necessary for successful BMT and that patients with DiGeorge syndrome with significant T cell deficiency may benefit from this treatment.

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Year:  1987        PMID: 3298595     DOI: 10.1016/s0022-3476(87)80339-6

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  12 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.  Immunologic reconstitution in 22q deletion (DiGeorge) syndrome.

Authors:  Sean A McGhee; Maria Garcia Lloret; E Richard Stiehm
Journal:  Immunol Res       Date:  2009       Impact factor: 2.829

Review 3.  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

4.  The DiGeorge sequence. II. Immunologic findings in partial and complete forms of the disorder.

Authors:  W Müller; H H Peter; H C Kallfelz; A Franz; C H Rieger
Journal:  Eur J Pediatr       Date:  1989-11       Impact factor: 3.183

Review 5.  Transplantation of fetal cells and tissue: an overview.

Authors:  A Fine
Journal:  CMAJ       Date:  1994-11-01       Impact factor: 8.262

Review 6.  Immunodeficiencies Associated with Abnormal Newborn Screening for T Cell and B Cell Lymphopenia.

Authors:  Soma Jyonouchi; Artemio M Jongco; Jennifer Puck; Kathleen E Sullivan
Journal:  J Clin Immunol       Date:  2017-03-28       Impact factor: 8.317

7.  Successful bone marrow transplantation with split lymphoid chimerism in DiGeorge syndrome.

Authors:  M S Borzy; D Ridgway; F J Noya; W T Shearer
Journal:  J Clin Immunol       Date:  1989-09       Impact factor: 8.317

8.  Post-natal ontogenesis of the T-cell receptor CD4 and CD8 Vbeta repertoire and immune function in children with DiGeorge syndrome.

Authors:  Caterina Cancrini; Maria Luisa Romiti; Andrea Finocchi; Silvia Di Cesare; Patrizia Ciaffi; Claudia Capponi; Savita Pahwa; Paolo Rossi
Journal:  J Clin Immunol       Date:  2005-05       Impact factor: 8.542

9.  Biased T-cell receptor repertoires in patients with chromosome 22q11.2 deletion syndrome (DiGeorge syndrome/velocardiofacial syndrome).

Authors:  M Pierdominici; F Mazzetta; E Caprini; M Marziali; M C Digilio; B Marino; A Aiuti; F Amati; G Russo; G Novelli; F Pandolfi; G Luzi; A Giovannetti
Journal:  Clin Exp Immunol       Date:  2003-05       Impact factor: 4.330

10.  Placental transfer of maternally-derived IgA precludes the use of guthrie card eluates as a screening tool for primary immunodeficiency diseases.

Authors:  Stephan Borte; Magdalena Janzi; Qiang Pan-Hammarström; Ulrika von Döbeln; Lennart Nordvall; Jacek Winiarski; Anders Fasth; Lennart Hammarström
Journal:  PLoS One       Date:  2012-08-16       Impact factor: 3.240

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