Literature DB >> 2788728

Prediction of persistent immunodeficiency in the DiGeorge anomaly.

J Bastian1, S Law, L Vogler, A Lawton, H Herrod, S Anderson, S Horowitz, R Hong.   

Abstract

To assess the natural history of the immune defect in DiGeorge anomaly, we reviewed serial immunologic studies in 18 patients. The diagnosis was made with criteria based on the concept of the DiGeorge anomaly as a field defect. Initial or early follow-up laboratory examination suggested moderate to normal T cell function in 14 patients. None of these patients have lost T cell capability; they have never had infections characteristic of T cell deficiency. Four patients had clinical and laboratory evidence of profound immunodeficiency. A decreased number of CD4+ cells (less than 400/microliters) and a decrease in phytohemagglutinin responsiveness (stimulation index less than 10) may be useful in discriminating patients with immunodeficiency; absolute lymphocyte count and immunoglobulin values were not informative. At the time of surgery, the thymus was not found in 11 of 14 patients; however, only two of these patients had immunodeficiency. Patients with a persistently low number of CD4+ cells and decreased phytohemagglutinin response are candidates for immunologic reconstitution.

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Year:  1989        PMID: 2788728     DOI: 10.1016/s0022-3476(89)80837-6

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


  26 in total

1.  Low thymic output in the 22q11.2 deletion syndrome measured by CCR9+CD45RA+ T cell counts and T cell receptor rearrangement excision circles.

Authors:  K Lima; T G Abrahamsen; I Foelling; S Natvig; L P Ryder; R W Olaussen
Journal:  Clin Exp Immunol       Date:  2010-05-10       Impact factor: 4.330

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.  Secondary immunologic consequences in chromosome 22q11.2 deletion syndrome (DiGeorge syndrome/velocardiofacial syndrome).

Authors:  R Zemble; E Luning Prak; K McDonald; D McDonald-McGinn; E Zackai; K Sullivan
Journal:  Clin Immunol       Date:  2010-05-15       Impact factor: 3.969

4.  The incidence of a deletion in chromosome 22Q11 in sporadic and familial conotruncal heart disease.

Authors:  K Devriendt; B Eyskens; A Swillen; M Dumoulin; M Gewillig; J P Fryns
Journal:  Eur J Pediatr       Date:  1996-08       Impact factor: 3.183

5.  Neonatal Levels of T-cell Receptor Excision Circles (TREC) in Patients with 22q11.2 Deletion Syndrome and Later Disease Features.

Authors:  Kiran A Gul; Torstein Øverland; Liv Osnes; Lars O Baumbusch; Rolf D Pettersen; Kari Lima; Tore G Abrahamsen
Journal:  J Clin Immunol       Date:  2015-03-27       Impact factor: 8.317

6.  Increased prevalence of immunoglobulin A deficiency in patients with the chromosome 22q11.2 deletion syndrome (DiGeorge syndrome/velocardiofacial syndrome).

Authors:  C A Smith; D A Driscoll; B S Emanuel; D M McDonald-McGinn; E H Zackai; K E Sullivan
Journal:  Clin Diagn Lab Immunol       Date:  1998-05

7.  Longitudinal analysis of lymphocyte function and numbers in the first year of life in chromosome 22q11.2 deletion syndrome (DiGeorge syndrome/velocardiofacial syndrome).

Authors:  K E Sullivan; D McDonald-McGinn; D A Driscoll; B S Emanuel; E H Zackai; A F Jawad
Journal:  Clin Diagn Lab Immunol       Date:  1999-11

Review 8.  Viral hepatitis.

Authors:  G V Gregorio; G Mieli-Vergani; A P Mowat
Journal:  Arch Dis Child       Date:  1994-04       Impact factor: 3.791

9.  Review of 54 patients with complete DiGeorge anomaly enrolled in protocols for thymus transplantation: outcome of 44 consecutive transplants.

Authors:  M Louise Markert; Blythe H Devlin; Marilyn J Alexieff; Jie Li; Elizabeth A McCarthy; Stephanie E Gupton; Ivan K Chinn; Laura P Hale; Thomas B Kepler; Min He; Marcella Sarzotti; Michael A Skinner; Henry E Rice; Jeffrey C Hoehner
Journal:  Blood       Date:  2007-02-06       Impact factor: 22.113

10.  Thymus transplantation restores the repertoires of forkhead box protein 3 (FoxP3)+ and FoxP3- T cells in complete DiGeorge anomaly.

Authors:  I K Chinn; J D Milner; P Scheinberg; D C Douek; M L Markert
Journal:  Clin Exp Immunol       Date:  2013-07       Impact factor: 4.330

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