Literature DB >> 10548584

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

K E Sullivan1, D McDonald-McGinn, D A Driscoll, B S Emanuel, E H Zackai, A F Jawad.   

Abstract

Chromosome 22q11.2 deletion syndrome is a common syndrome typically consisting of variable cardiac defects, hypoparathyroidism, developmental delay, and immunodeficiency. The hemizygous deletion has variable effects on the immune system even within the same kindred, and the extent of the immunodeficiency is difficult to predict. Some patients have shown improvement over time; however, this is the first prospective longitudinal study of the dynamic nature of the immunodeficiency. Nineteen patients were studied prospectively between 1994 and 1997. The results of the newborn immunologic studies in the chromosome 22q11.2 deletion group were significantly different from those of a group of newborns with cardiac disease due to other causes. Peripheral blood T-cell numbers were decreased in the chromosome 22q11.2 deletion group, although T-cell function was largely preserved. The group as a whole demonstrated few changes in the first year of life, but a subset of patients with markedly diminished T-cell numbers did demonstrate improvement. Therefore, improvement in peripheral blood T-cell counts is variable in chromosome 22q11.2 deletion syndrome. The patients with the lowest T-cell counts improved the most in the first year of life.

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Year:  1999        PMID: 10548584      PMCID: PMC95796          DOI: 10.1128/CDLI.6.6.906-911.1999

Source DB:  PubMed          Journal:  Clin Diagn Lab Immunol        ISSN: 1071-412X


  48 in total

1.  Molecular definition of 22q11 deletions in 151 velo-cardio-facial syndrome patients.

Authors:  C Carlson; H Sirotkin; R Pandita; R Goldberg; J McKie; R Wadey; S R Patanjali; S M Weissman; K Anyane-Yeboa; D Warburton; P Scambler; R Shprintzen; R Kucherlapati; B E Morrow
Journal:  Am J Hum Genet       Date:  1997-09       Impact factor: 11.025

2.  The human thymus contains a novel population of B lymphocytes.

Authors:  P G Isaacson; A J Norton; B J Addis
Journal:  Lancet       Date:  1987-12-26       Impact factor: 79.321

3.  T-cell deficiency in diGeorge syndrome.

Authors:  H W Lischner; D S Huff
Journal:  Birth Defects Orig Artic Ser       Date:  1975

4.  T-cell subsets and natural killer cells in DiGeorge and SCID patients.

Authors:  M C Sirianni; L Businco; L Fiore; R Seminara; F Aiuti
Journal:  Birth Defects Orig Artic Ser       Date:  1983

5.  DiGeorge syndrome: long-term survival complicated by Graves disease.

Authors:  A J Ham Pong; A Cavallo; G H Holman; A S Goldman
Journal:  J Pediatr       Date:  1985-04       Impact factor: 4.406

Review 6.  The DiGeorge syndrome. I. Clinical evaluation and course of partial and complete forms of the syndrome.

Authors:  W Müller; H H Peter; M Wilken; H Jüppner; H C Kallfelz; H P Krohn; K Miller; C H Rieger
Journal:  Eur J Pediatr       Date:  1988-06       Impact factor: 3.183

7.  A deletion in chromosome 22 can cause DiGeorge syndrome.

Authors:  A de la Chapelle; R Herva; M Koivisto; P Aula
Journal:  Hum Genet       Date:  1981       Impact factor: 4.132

8.  The association of the DiGeorge anomalad with partial monosomy of chromosome 22.

Authors:  R I Kelley; E H Zackai; B S Emanuel; M Kistenmacher; F Greenberg; H H Punnett
Journal:  J Pediatr       Date:  1982-08       Impact factor: 4.406

9.  The spectrum of the DiGeorge syndrome.

Authors:  M E Conley; J B Beckwith; J F Mancer; L Tenckhoff
Journal:  J Pediatr       Date:  1979-06       Impact factor: 4.406

10.  Severe combined immunodeficiencies, primary T-cell defects and DiGeorge syndrome in humans: characterization by monoclonal antibodies and natural killer cell activity.

Authors:  M C Sirianni; L Businco; R Seminara; F Aiuti
Journal:  Clin Immunol Immunopathol       Date:  1983-09
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  20 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.  Live viral vaccines in a DiGeorge syndrome patient.

Authors:  V Waters; K S Peterson; P LaRussa
Journal:  Arch Dis Child       Date:  2006-06-23       Impact factor: 3.791

Review 3.  Immunological aspects of 22q11.2 deletion syndrome.

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

4.  Biochemical characterisation of the proteins encoded by the DiGeorge critical region 6 (DGCR6) genes.

Authors:  Thorsten Pfuhl; Matthias Dürr; Andreas Spurk; Björn Schwalbert; Ruth Nord; Josef Mysliwietz; Elisabeth Kremmer; Friedrich A Grässer
Journal:  Hum Genet       Date:  2005-04-09       Impact factor: 4.132

Review 5.  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

6.  Multiplexed quantitative real-time PCR to detect 22q11.2 deletion in patients with congenital heart disease.

Authors:  Aoy Tomita-Mitchell; Donna K Mahnke; Joshua M Larson; Sujana Ghanta; Ying Feng; Pippa M Simpson; Ulrich Broeckel; Kelly Duffy; James S Tweddell; William J Grossman; John M Routes; Michael E Mitchell
Journal:  Physiol Genomics       Date:  2010-06-15       Impact factor: 3.107

7.  Persistent low thymic activity and non-cardiac mortality in children with chromosome 22q11.2 microdeletion and partial DiGeorge syndrome.

Authors:  P Eberle; C Berger; S Junge; S Dougoud; E Valsangiacomo Büchel; M Riegel; A Schinzel; R Seger; T Güngör
Journal:  Clin Exp Immunol       Date:  2008-11-24       Impact factor: 4.330

8.  CHARGE (coloboma, heart defect, atresia choanae, retarded growth and development, genital hypoplasia, ear anomalies/deafness) syndrome and chromosome 22q11.2 deletion syndrome: a comparison of immunologic and nonimmunologic phenotypic features.

Authors:  Soma Jyonouchi; Donna M McDonald-McGinn; Sherri Bale; Elaine H Zackai; Kathleen E Sullivan
Journal:  Pediatrics       Date:  2009-05       Impact factor: 7.124

Review 9.  22q11.2 deletion syndrome.

Authors:  Donna M McDonald-McGinn; Kathleen E Sullivan; Bruno Marino; Nicole Philip; Ann Swillen; Jacob A S Vorstman; Elaine H Zackai; Beverly S Emanuel; Joris R Vermeesch; Bernice E Morrow; Peter J Scambler; Anne S Bassett
Journal:  Nat Rev Dis Primers       Date:  2015-11-19       Impact factor: 52.329

10.  Maturational alterations of peripheral T cell subsets and cytokine gene expression in 22q11.2 deletion syndrome.

Authors:  Y Kanaya; S Ohga; K Ikeda; K Furuno; T Ohno; H Takada; N Kinukawa; T Hara
Journal:  Clin Exp Immunol       Date:  2006-04       Impact factor: 4.330

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