Literature DB >> 29341423

Variable immune deficiency related to deletion size in chromosome 22q11.2 deletion syndrome.

Blaine Crowley1, Melanie Ruffner2, Donna M McDonald McGinn1, Kathleen E Sullivan2.   

Abstract

The clinical features of 22q11.2 deletion syndrome include virtually every organ of the body. This review will focus on the immune system and the differences related to deletion breakpoints. A hypoplastic thymus was one of the first features described in this syndrome and low T cell counts, as a consequence of thymic hypoplasia, are the most commonly described immunologic feature. These are most prominently seen in early childhood and can be associated with increased persistence of viruses. Later in life, evidence of T cell exhaustion may be seen and secondary deficiencies of antibody function have been described. The relationship of the immunodeficiency to the deletion breakpoints has been understudied due to the infrequent analysis of people carrying smaller deletions. This manuscript will review the immune deficiency in 22q11.2 deletion syndrome and describe differences in the T cell counts related to the deletion breakpoints. Distal, non-TBX1 inclusive deletions, were found to be associated with better T cell counts. Another new finding is the relative preservation of T cell counts in those patients with a 22q11.2 duplication.
© 2018 Wiley Periodicals, Inc.

Entities:  

Keywords:  DiGeorge; IgG; T cells; breakpoints; deletion size; low copy number repeats

Mesh:

Year:  2018        PMID: 29341423      PMCID: PMC6470357          DOI: 10.1002/ajmg.a.38597

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  55 in total

1.  Screening for celiac disease in patients with deletion 22q11.2 (DiGeorge/velo-cardio-facial syndrome).

Authors:  Maria Cristina Digilio; Aldo Giannotti; Massimo Castro; Franco Colistro; Francesca Ferretti; Bruno Marino; Bruno Dallapiccola
Journal:  Am J Med Genet A       Date:  2003-09-01       Impact factor: 2.802

Review 2.  Lymphodepletion and homeostatic proliferation: implications for transplantation.

Authors:  N K Tchao; L A Turka
Journal:  Am J Transplant       Date:  2012-03-15       Impact factor: 8.086

3.  B cell development in chromosome 22q11.2 deletion syndrome.

Authors:  Beata Derfalvi; Kelly Maurer; Donna M McDonald McGinn; Elaine Zackai; Wenzhao Meng; Eline T Luning Prak; Kathleen E Sullivan
Journal:  Clin Immunol       Date:  2015-12-10       Impact factor: 3.969

4.  Evaluation of potential modifiers of the palatal phenotype in the 22q11.2 deletion syndrome.

Authors:  Deborah A Driscoll; Torrey Boland; Beverly S Emanuel; Richard E Kirschner; Don LaRossa; Jeanne Manson; Donna McDonald-McGinn; Peter Randall; Cynthia Solot; Elaine Zackai; Laura E Mitchell
Journal:  Cleft Palate Craniofac J       Date:  2006-07

5.  Clinical features of 78 adults with 22q11 Deletion Syndrome.

Authors:  Anne S Bassett; Eva W C Chow; Janice Husted; Rosanna Weksberg; Oana Caluseriu; Gary D Webb; Michael A Gatzoulis
Journal:  Am J Med Genet A       Date:  2005-11-01       Impact factor: 2.802

6.  Rare Genome-Wide Copy Number Variation and Expression of Schizophrenia in 22q11.2 Deletion Syndrome.

Authors:  Anne S Bassett; Chelsea Lowther; Daniele Merico; Gregory Costain; Eva W C Chow; Therese van Amelsvoort; Donna McDonald-McGinn; Raquel E Gur; Ann Swillen; Marianne Van den Bree; Kieran Murphy; Doron Gothelf; Carrie E Bearden; Stephan Eliez; Wendy Kates; Nicole Philip; Vandana Sashi; Linda Campbell; Jacob Vorstman; Joseph Cubells; Gabriela M Repetto; Tony Simon; Erik Boot; Tracy Heung; Rens Evers; Claudia Vingerhoets; Esther van Duin; Elaine Zackai; Elfi Vergaelen; Koen Devriendt; Joris R Vermeesch; Michael Owen; Clodagh Murphy; Elena Michaelovosky; Leila Kushan; Maude Schneider; Wanda Fremont; Tiffany Busa; Stephen Hooper; Kathryn McCabe; Sasja Duijff; Karin Isaev; Giovanna Pellecchia; John Wei; Matthew J Gazzellone; Stephen W Scherer; Beverly S Emanuel; Tingwei Guo; Bernice E Morrow; Christian R Marshall
Journal:  Am J Psychiatry       Date:  2017-07-28       Impact factor: 18.112

7.  Reduction of CRKL expression in patients with partial DiGeorge syndrome is associated with impairment of T-cell functions.

Authors:  Mauro Giacomelli; Rajesh Kumar; Annarosa Soresina; Nicola Tamassia; Tiziana Lorenzini; Daniele Moratto; Sara Gasperini; Marco Cassatella; Alessandro Plebani; Vassilios Lougaris; Raffaele Badolato
Journal:  J Allergy Clin Immunol       Date:  2016-02-11       Impact factor: 10.793

8.  Association between hypoparathyroidism and defective T cell immunity in 22q11.2 deletion syndrome.

Authors:  A Herwadkar; A R Gennery; A S Moran; M R Haeney; P D Arkwright
Journal:  J Clin Pathol       Date:  2010-02       Impact factor: 3.411

9.  Allergies in patients with chromosome 22q11.2 deletion syndrome (DiGeorge syndrome/velocardiofacial syndrome) and patients with chronic granulomatous disease.

Authors:  Lauren Staple; Timothy Andrews; Donna McDonald-McGinn; Elaine Zackai; Kathleen E Sullivan
Journal:  Pediatr Allergy Immunol       Date:  2005-05       Impact factor: 6.377

10.  Low marginal zone-like B lymphocytes and natural antibodies characterize skewed B-lymphocyte subpopulations in del22q11 DiGeorge patients.

Authors:  Adam Klocperk; Ester Mejstříková; Jana Kayserová; Tomáš Kalina; Anna Šedivá
Journal:  Clin Immunol       Date:  2015-09-02       Impact factor: 3.969

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  16 in total

1.  Lymphocyte Apoptosis and FAS Expression in Patients with 22q11.2 Deletion Syndrome.

Authors:  Dina M Aresvik; Torstein Øverland; Kari Lima; Rolf D Pettersen; Tore G Abrahamsen
Journal:  J Clin Immunol       Date:  2018-12-19       Impact factor: 8.317

Review 2.  [Ectopic thymic tissue and ectopic thymic tumors].

Authors:  A Marx; T Rüdiger; E Rößner; A Tzankov; V T de Montpréville; R R Rieker; P Ströbel; C-A Weis
Journal:  Pathologe       Date:  2018-09       Impact factor: 1.011

3.  Vitamin D status and the immune assessment in 22q11.2 deletion syndrome.

Authors:  A Legitimo; V Bertini; G Costagliola; G I Baroncelli; R Morganti; A Valetto; R Consolini
Journal:  Clin Exp Immunol       Date:  2020-04-16       Impact factor: 4.330

Review 4.  Immune and Genetic Features of the Chromosome 22q11.2 Deletion (DiGeorge Syndrome).

Authors:  Caroline Y Kuo; Rebecca Signer; Sulagna C Saitta
Journal:  Curr Allergy Asthma Rep       Date:  2018-10-30       Impact factor: 4.806

5.  Unexpected combination: DiGeorge syndrome and myeloperoxidase deficiency.

Authors:  Simona Abraitytė; Elisabeth Kotsi; Lisa Anne Devlin; John David Moore Edgar
Journal:  BMJ Case Rep       Date:  2020-02-26

6.  Relationship Between Severity of T Cell Lymphopenia and Immune Dysregulation in Patients with DiGeorge Syndrome (22q11.2 Deletions and/or Related TBX1 Mutations): a USIDNET Study.

Authors:  Deepti R Deshpande; Yesim Y Demirdag; Rebecca A Marsh; Kathleen E Sullivan; Jordan S Orange
Journal:  J Clin Immunol       Date:  2020-09-19       Impact factor: 8.317

7.  Testing criteria for 22q11.2 deletion syndrome: preliminary results of a low cost strategy for public health.

Authors:  Ilária Cristina Sgardioli; Fabíola Paoli Monteiro; Paulo Fanti; Társis Paiva Vieira; Vera Lúcia Gil-da-Silva-Lopes
Journal:  Orphanet J Rare Dis       Date:  2019-06-03       Impact factor: 4.123

8.  Thymic Epithelium Abnormalities in DiGeorge and Down Syndrome Patients Contribute to Dysregulation in T Cell Development.

Authors:  Genni Enza Marcovecchio; Ileana Bortolomai; Francesca Ferrua; Elena Fontana; Luisa Imberti; Erika Conforti; Donato Amodio; Sonia Bergante; Giulia Macchiarulo; Veronica D'Oria; Francesca Conti; Silvia Di Cesare; Georgia Fousteri; Adriano Carotti; Alessandro Giamberti; Pietro Luigi Poliani; Luigi D Notarangelo; Caterina Cancrini; Anna Villa; Marita Bosticardo
Journal:  Front Immunol       Date:  2019-03-15       Impact factor: 7.561

Review 9.  Inborn errors of thymic stromal cell development and function.

Authors:  Alexandra Y Kreins; Stefano Maio; Fatima Dhalla
Journal:  Semin Immunopathol       Date:  2020-11-30       Impact factor: 9.623

10.  Candidate modifier genes for immune function in 22q11.2 deletion syndrome.

Authors:  Catherina T Pinnaro; Travis Henry; Heather J Major; Mrutyunjaya Parida; Lucy E DesJardin; John R Manak; Benjamin W Darbro
Journal:  Mol Genet Genomic Med       Date:  2019-12-12       Impact factor: 2.183

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