Literature DB >> 31468084

Familial hypogammaglobulinemia with high RTE and naïve T lymphocytes.

Elisa Piscianz1, Ester Conversano2, Anna Monica Bianco3, Flavio Faletra3, Alberto Tommasini3, Erica Valencic3.   

Abstract

Most of primary immunodeficiencies with hypogammaglobulinemia are associated with reduced memory B cells. T cell development may be interesting as well, but increased recent thymic emigrants are rarely reported in these patients. We report the case of a family (mother and her two sons) diagnosed with common variable immunodeficiency 10 due to a mutation in the NFKB2 gene. Laboratory findings showed that all three patients presented hypogammaglobulinemia, reduced memory B cells and elevated naïve T lymphocytes and recent thymic emigrants. This feature, in the absence of glucocorticoid deficiency, may suggest a primary thymic dysfunction. Interestingly, the mother presented the worst immune phenotype, as regards both antibody production and NK function, indicating that immune function may deteriorate in the course of time. We conclude that close monitoring of immune functions may widen the knowledge on the CVID10 and improve the patients' care.

Entities:  

Keywords:  Autoimmunity; Common variable immunodeficiency; NFKB2; NK degranulation; Recent thymic emigrants

Mesh:

Substances:

Year:  2019        PMID: 31468084     DOI: 10.1007/s00011-019-01277-1

Source DB:  PubMed          Journal:  Inflamm Res        ISSN: 1023-3830            Impact factor:   4.575


  6 in total

1.  Defective natural killer-cell cytotoxic activity in NFKB2-mutated CVID-like disease.

Authors:  Vassilios Lougaris; Giovanna Tabellini; Massimiliano Vitali; Manuela Baronio; Ornella Patrizi; Giacomo Tampella; Augusto Biasini; Daniele Moratto; Silvia Parolini; Alessandro Plebani
Journal:  J Allergy Clin Immunol       Date:  2015-01-17       Impact factor: 10.793

2.  Functional evaluation of natural killer cell cytotoxic activity in NFKB2-mutated patients.

Authors:  Davide Montin; Francesco Licciardi; Elisa Giorgio; Andrea Ciolfi; Simone Pizzi; Alessandro Mussa; Raffaella Meazza; Marco Tartaglia; Alfredo Brusco; Daniela Pende; Giovanni Battista Ferrero
Journal:  Immunol Lett       Date:  2017-12-24       Impact factor: 3.685

3.  Autosomal-dominant B-cell deficiency with alopecia due to a mutation in NFKB2 that results in nonprocessable p100.

Authors:  Cindy Eunhee Lee; David A Fulcher; Belinda Whittle; Rochna Chand; Nicole Fewings; Matthew Field; Daniel Andrews; Christopher C Goodnow; Matthew C Cook
Journal:  Blood       Date:  2014-09-18       Impact factor: 22.113

Review 4.  NF-κB: roles and regulation in different CD4(+) T-cell subsets.

Authors:  Hyunju Oh; Sankar Ghosh
Journal:  Immunol Rev       Date:  2013-03       Impact factor: 12.988

5.  Germline mutations in NFKB2 implicate the noncanonical NF-κB pathway in the pathogenesis of common variable immunodeficiency.

Authors:  Karin Chen; Emily M Coonrod; Attila Kumánovics; Zechariah F Franks; Jacob D Durtschi; Rebecca L Margraf; Wilfred Wu; Nahla M Heikal; Nancy H Augustine; Perry G Ridge; Harry R Hill; Lynn B Jorde; Andrew S Weyrich; Guy A Zimmerman; Adi V Gundlapalli; John F Bohnsack; Karl V Voelkerding
Journal:  Am J Hum Genet       Date:  2013-10-17       Impact factor: 11.025

6.  Mutations in NFKB2 and potential genetic heterogeneity in patients with DAVID syndrome, having variable endocrine and immune deficiencies.

Authors:  Thierry Brue; Marie-Hélène Quentien; Konstantin Khetchoumian; Marco Bensa; José-Mario Capo-Chichi; Brigitte Delemer; Aurelio Balsalobre; Christina Nassif; Dimitris T Papadimitriou; Anne Pagnier; Caroline Hasselmann; Lysanne Patry; Jeremy Schwartzentruber; Pierre-François Souchon; Shinobu Takayasu; Alain Enjalbert; Guy Van Vliet; Jacek Majewski; Jacques Drouin; Mark E Samuels
Journal:  BMC Med Genet       Date:  2014-12-19       Impact factor: 2.103

  6 in total
  1 in total

1.  Immunity and Genetics at the Revolving Doors of Diagnostics in Primary Immunodeficiencies.

Authors:  Francesco Rispoli; Erica Valencic; Martina Girardelli; Alessia Pin; Alessandra Tesser; Elisa Piscianz; Valentina Boz; Flavio Faletra; Giovanni Maria Severini; Andrea Taddio; Alberto Tommasini
Journal:  Diagnostics (Basel)       Date:  2021-03-16
  1 in total

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