Literature DB >> 25216719

Immunodeficiency associated with a nonsense mutation of IKBKB.

Christian Nielsen1, Marianne A Jakobsen, Martin Jakob Larsen, Amanda C Müller, Soren Hansen, Søren T Lillevang, Niels Fisker, Torben Barington.   

Abstract

We report an infant of consanguineous parents of Turkish decent with a novel immunodeficiency associated with homozygosity for a nonsense mutation of the gene encoding Inhibitor of nuclear factor kappa-B (NF-κB) kinase subunit beta (IKKβ). At five months, she presented with respiratory insufficiency and Pneumocystis jirovecii pneumonia which was successfully treated. At nine months, iatrogenic systemic infection with Mycobacterium bovis was found and eventually led to her death at age 14 months. Laboratory findings were reminiscent of hyper-IgM syndrome, but genetic testing gave no explanation before whole exome sequencing revealed a novel mutation abrogating signaling through the canonical NF-κB pathway.

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Year:  2014        PMID: 25216719     DOI: 10.1007/s10875-014-0097-1

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.317


  19 in total

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Authors:  Andrea Oeckinghaus; Matthew S Hayden; Sankar Ghosh
Journal:  Nat Immunol       Date:  2011-07-19       Impact factor: 25.606

2.  Recombinant interleukin 2 therapy in severe combined immunodeficiency disease.

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3.  From FastQ data to high confidence variant calls: the Genome Analysis Toolkit best practices pipeline.

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Journal:  Curr Protoc Bioinformatics       Date:  2013

4.  A novel X-linked disorder of immune deficiency and hypohidrotic ectodermal dysplasia is allelic to incontinentia pigmenti and due to mutations in IKK-gamma (NEMO).

Authors:  J Zonana; M E Elder; L C Schneider; S J Orlow; C Moss; M Golabi; S K Shapira; P A Farndon; D W Wara; S A Emmal; B M Ferguson
Journal:  Am J Hum Genet       Date:  2000-10-24       Impact factor: 11.025

5.  Mutant CHUK and severe fetal encasement malformation.

Authors:  Jenni Lahtela; Heidi O Nousiainen; Vedran Stefanovic; Jonna Tallila; Heli Viskari; Riitta Karikoski; Massimiliano Gentile; Carola Saloranta; Teppo Varilo; Riitta Salonen; Marjo Kestilä
Journal:  N Engl J Med       Date:  2010-10-21       Impact factor: 91.245

6.  Mature T cells depend on signaling through the IKK complex.

Authors:  Marc Schmidt-Supprian; Gilles Courtois; Jane Tian; Anthony J Coyle; Alain Israël; Klaus Rajewsky; Manolis Pasparakis
Journal:  Immunity       Date:  2003-09       Impact factor: 31.745

7.  Deficiency of innate and acquired immunity caused by an IKBKB mutation.

Authors:  Ulrich Pannicke; Bernd Baumann; Sebastian Fuchs; Philipp Henneke; Anne Rensing-Ehl; Marta Rizzi; Ales Janda; Katrin Hese; Michael Schlesier; Karlheinz Holzmann; Stephan Borte; Constanze Laux; Eva-Maria Rump; Alan Rosenberg; Teresa Zelinski; Hubert Schrezenmeier; Thomas Wirth; Stephan Ehl; Marlis L Schroeder; Klaus Schwarz
Journal:  N Engl J Med       Date:  2013-12-26       Impact factor: 91.245

8.  A novel gain-of-function IKBA mutation underlies ectodermal dysplasia with immunodeficiency and polyendocrinopathy.

Authors:  Lena F Schimke; Nikolaus Rieber; Stacey Rylaarsdam; Otávio Cabral-Marques; Nicholas Hubbard; Anne Puel; Laura Kallmann; Stephanie Anover Sombke; Gundula Notheis; Hans-Peter Schwarz; Birgit Kammer; Tomas Hökfelt; Reinald Repp; Capucine Picard; Jean-Laurent Casanova; Bernd H Belohradsky; Michael H Albert; Hans D Ochs; Ellen D Renner; Troy R Torgerson
Journal:  J Clin Immunol       Date:  2013-05-25       Impact factor: 8.317

9.  A hypermorphic IkappaBalpha mutation is associated with autosomal dominant anhidrotic ectodermal dysplasia and T cell immunodeficiency.

Authors:  Gilles Courtois; Asma Smahi; Janine Reichenbach; Rainer Döffinger; Caterina Cancrini; Marion Bonnet; Anne Puel; Christine Chable-Bessia; Shoji Yamaoka; Jacqueline Feinberg; Sophie Dupuis-Girod; Christine Bodemer; Susanna Livadiotti; Francesco Novelli; Paolo Rossi; Alain Fischer; Alain Israël; Arnold Munnich; Françoise Le Deist; Jean-Laurent Casanova
Journal:  J Clin Invest       Date:  2003-10       Impact factor: 14.808

10.  The IKKbeta subunit of IkappaB kinase (IKK) is essential for nuclear factor kappaB activation and prevention of apoptosis.

Authors:  Z W Li; W Chu; Y Hu; M Delhase; T Deerinck; M Ellisman; R Johnson; M Karin
Journal:  J Exp Med       Date:  1999-06-07       Impact factor: 14.307

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  20 in total

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2.  Identification of genes associated with susceptibility to Mycobacterium avium ssp. paratuberculosis (Map) tissue infection in Holstein cattle using gene set enrichment analysis-SNP.

Authors:  J N Kiser; M Neupane; S N White; H L Neibergs
Journal:  Mamm Genome       Date:  2017-11-28       Impact factor: 2.957

3.  Newborn Screening for IKBKB Deficiency in Manitoba, Using Genetic Mutation Analysis.

Authors:  Tamar S Rubin; Cheryl Rockman-Greenberg; Paul Van Caeseele; Geoffrey D E Cuvelier; Luvinia Kwan; Marlis L Schroeder
Journal:  J Clin Immunol       Date:  2018-10-04       Impact factor: 8.317

Review 4.  The role of monogenic disease in children with very early onset inflammatory bowel disease.

Authors:  Judith R Kelsen; Robert N Baldassano
Journal:  Curr Opin Pediatr       Date:  2017-10       Impact factor: 2.856

Review 5.  Genomic and Immunologic Drivers of Very Early-Onset Inflammatory Bowel Disease.

Authors:  Maire A Conrad; Judith R Kelsen
Journal:  Pediatr Dev Pathol       Date:  2019-03-06

Review 6.  IKK-related genetic diseases: probing NF-κB functions in humans and other matters.

Authors:  Anna Senegas; Jérémie Gautheron; Alice Gentil Dit Maurin; Gilles Courtois
Journal:  Cell Mol Life Sci       Date:  2014-11-29       Impact factor: 9.261

7.  T Cell Impairment Is Predictive for a Severe Clinical Course in NEMO Deficiency.

Authors:  Stephanie Heller; Uwe Kölsch; Thomas Magg; Renate Krüger; Andrea Scheuern; Holm Schneider; Anna Eichinger; Volker Wahn; Nadine Unterwalder; Myriam Lorenz; Klaus Schwarz; Christian Meisel; Ansgar Schulz; Fabian Hauck; Horst von Bernuth
Journal:  J Clin Immunol       Date:  2020-01-21       Impact factor: 8.317

Review 8.  The Child with Recurrent Mycobacterial Disease.

Authors:  Brian Reed; William K Dolen
Journal:  Curr Allergy Asthma Rep       Date:  2018-06-23       Impact factor: 4.806

9.  Maintaining intestinal health: the genetics and immunology of very early onset inflammatory bowel disease.

Authors:  Judith R Kelsen; Robert N Baldassano; David Artis; Gregory F Sonnenberg
Journal:  Cell Mol Gastroenterol Hepatol       Date:  2015-09-01

10.  Human HOIP and LUBAC deficiency underlies autoinflammation, immunodeficiency, amylopectinosis, and lymphangiectasia.

Authors:  Bertrand Boisson; Emmanuel Laplantine; Kerry Dobbs; Aurélie Cobat; Nadine Tarantino; Melissa Hazen; Hart G W Lidov; Gregory Hopkins; Likun Du; Aziz Belkadi; Maya Chrabieh; Yuval Itan; Capucine Picard; Jean-Christophe Fournet; Hermann Eibel; Erdyni Tsitsikov; Sung-Yun Pai; Laurent Abel; Waleed Al-Herz; Jean-Laurent Casanova; Alain Israel; Luigi D Notarangelo
Journal:  J Exp Med       Date:  2015-05-25       Impact factor: 14.307

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