Literature DB >> 25104208

Atypical severe combined immunodeficiency caused by a novel homozygous mutation in Rag1 gene in a girl who presented with pyoderma gangrenosum: a case report and literature review.

Turkan Patiroglu1, H Haluk Akar, Kimberly Gilmour, M Akif Ozdemir, Shahnaz Bibi, Frances Henriquez, Siobhan O Burns, Ekrem Unal.   

Abstract

Severe combined immunodeficiency (SCID) is a heterogeneous group of inherited defects involving the development of T- and/or B-lymphocytes. We report a female with atypical severe combined immunodeficiency caused by a novel homozygous mutation at cDNA position 2290 (c.2290C > T) in exon 2 of the RAG1 gene. The patient presented with bronchopneumonia, pyoderma gangrenosum (PG), pancytopenia and splenomegaly. She presented to us with pancytopenia and splenomegaly at the age of 11. Her condition was complicated by PG on left lower ankle at the age of 12. She experienced bronchopneumonia at the age of 15. She was diagnosed with RAG1 deficiency at the age of 16. Her immunological presentation included leucopenia and diminished number of B cells.

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Year:  2014        PMID: 25104208     DOI: 10.1007/s10875-014-0077-5

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


  21 in total

1.  Have we seen the last variant of severe combined immunodeficiency?

Authors:  Alain Fischer
Journal:  N Engl J Med       Date:  2003-11-06       Impact factor: 91.245

Review 2.  Clinical and immunological manifestations of patients with atypical severe combined immunodeficiency.

Authors:  Kerstin Felgentreff; Ruy Perez-Becker; Carsten Speckmann; Klaus Schwarz; Krzysztof Kalwak; Gasper Markelj; Tadej Avcin; Waseem Qasim; E G Davies; Tim Niehues; Stephan Ehl
Journal:  Clin Immunol       Date:  2011-05-30       Impact factor: 3.969

Review 3.  RAG-dependent primary immunodeficiencies.

Authors:  Cristina Sobacchi; Veronica Marrella; Francesca Rucci; Paolo Vezzoni; Anna Villa
Journal:  Hum Mutat       Date:  2006-12       Impact factor: 4.878

4.  RAG mutations in human B cell-negative SCID.

Authors:  K Schwarz; G H Gauss; L Ludwig; U Pannicke; Z Li; D Lindner; W Friedrich; R A Seger; T E Hansen-Hagge; S Desiderio; M R Lieber; C R Bartram
Journal:  Science       Date:  1996-10-04       Impact factor: 47.728

5.  Expanding the spectrum of recombination-activating gene 1 deficiency: a family with early-onset autoimmunity.

Authors:  Lauren A Henderson; Francesco Frugoni; Gregory Hopkins; Helen de Boer; Sung-Yun Pai; Yu Nee Lee; Jolan E Walter; Melissa M Hazen; Luigi D Notarangelo
Journal:  J Allergy Clin Immunol       Date:  2013-07-24       Impact factor: 10.793

6.  Leaky severe combined immunodeficiency and aberrant DNA rearrangements due to a hypomorphic RAG1 mutation.

Authors:  William Giblin; Monalisa Chatterji; Gerwin Westfield; Tehmina Masud; Brian Theisen; Hwei-Ling Cheng; Jeffrey DeVido; Frederick W Alt; David O Ferguson; David G Schatz; JoAnn Sekiguchi
Journal:  Blood       Date:  2009-01-06       Impact factor: 22.113

7.  Novel mutatıons and diverse clinical phenotypes in recombinase-activating gene 1 deficiency.

Authors:  Necil Kutukculer; Nesrin Gulez; Neslihan Edeer Karaca; Guzide Aksu; Afig Berdeli
Journal:  Ital J Pediatr       Date:  2012-03-16       Impact factor: 2.638

8.  A novel immunodeficiency associated with hypomorphic RAG1 mutations and CMV infection.

Authors:  Jean-Pierre de Villartay; Annick Lim; Hamoud Al-Mousa; Sophie Dupont; Julie Déchanet-Merville; Edith Coumau-Gatbois; Marie-Lise Gougeon; Arnaud Lemainque; Céline Eidenschenk; Emmanuelle Jouanguy; Laurent Abel; Jean-Laurent Casanova; Alain Fischer; Françoise Le Deist
Journal:  J Clin Invest       Date:  2005-11       Impact factor: 14.808

9.  A systematic analysis of recombination activity and genotype-phenotype correlation in human recombination-activating gene 1 deficiency.

Authors:  Yu Nee Lee; Francesco Frugoni; Kerry Dobbs; Jolan E Walter; Silvia Giliani; Andrew R Gennery; Waleed Al-Herz; Elie Haddad; Francoise LeDeist; Jack H Bleesing; Lauren A Henderson; Sung-Yun Pai; Robert P Nelson; Dalia H El-Ghoneimy; Reem A El-Feky; Shereen M Reda; Elham Hossny; Pere Soler-Palacin; Ramsay L Fuleihan; Niraj C Patel; Michel J Massaad; Raif S Geha; Jennifer M Puck; Paolo Palma; Caterina Cancrini; Karin Chen; Mauno Vihinen; Frederick W Alt; Luigi D Notarangelo
Journal:  J Allergy Clin Immunol       Date:  2013-11-28       Impact factor: 10.793

Review 10.  Screening for severe combined immunodeficiency in neonates.

Authors:  Brian T Kelly; Jonathan S Tam; James W Verbsky; John M Routes
Journal:  Clin Epidemiol       Date:  2013-09-16       Impact factor: 4.790

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  10 in total

Review 1.  History and current status of newborn screening for severe combined immunodeficiency.

Authors:  Antonia Kwan; Jennifer M Puck
Journal:  Semin Perinatol       Date:  2015-04-30       Impact factor: 3.300

2.  T+ NK+ IL-2 Receptor γ Chain Mutation: a Challenging Diagnosis of Atypical Severe Combined Immunodeficiency.

Authors:  Polina Stepensky; Baerbel Keller; Oded Shamriz; Caroline von Spee-Mayer; David Friedmann; Bella Shadur; Susanne Unger; Sebastian Fuchs; Adeeb NaserEddin; Nisreen Rumman; Sara Amro; Vered Molho Pessach; Omar Abuzaitoun; Raz Somech; Orly Elpeleg; Stephan Ehl; Klaus Warnatz
Journal:  J Clin Immunol       Date:  2018-06-09       Impact factor: 8.317

3.  Characterization of T and B cell repertoire diversity in patients with RAG deficiency.

Authors:  Yu Nee Lee; Francesco Frugoni; Kerry Dobbs; Irit Tirosh; Likun Du; Francesca A Ververs; Heng Ru; Lisa Ott de Bruin; Mehdi Adeli; Jacob H Bleesing; David Buchbinder; Manish J Butte; Caterina Cancrini; Karin Chen; Sharon Choo; Reem A Elfeky; Andrea Finocchi; Ramsay L Fuleihan; Andrew R Gennery; Dalia H El-Ghoneimy; Lauren A Henderson; Waleed Al-Herz; Elham Hossny; Robert P Nelson; Sung-Yun Pai; Niraj C Patel; Shereen M Reda; Pere Soler-Palacin; Raz Somech; Paolo Palma; Hao Wu; Silvia Giliani; Jolan E Walter; Luigi D Notarangelo
Journal:  Sci Immunol       Date:  2016-12-16

Review 4.  Human RAG mutations: biochemistry and clinical implications.

Authors:  Luigi D Notarangelo; Min-Sung Kim; Jolan E Walter; Yu Nee Lee
Journal:  Nat Rev Immunol       Date:  2016-03-21       Impact factor: 53.106

5.  Broad-spectrum antibodies against self-antigens and cytokines in RAG deficiency.

Authors:  Jolan E Walter; Lindsey B Rosen; Krisztian Csomos; Jacob M Rosenberg; Divij Mathew; Marton Keszei; Boglarka Ujhazi; Karin Chen; Yu Nee Lee; Irit Tirosh; Kerry Dobbs; Waleed Al-Herz; Morton J Cowan; Jennifer Puck; Jack J Bleesing; Michael S Grimley; Harry Malech; Suk See De Ravin; Andrew R Gennery; Roshini S Abraham; Avni Y Joshi; Thomas G Boyce; Manish J Butte; Kari C Nadeau; Imelda Balboni; Kathleen E Sullivan; Javeed Akhter; Mehdi Adeli; Reem A El-Feky; Dalia H El-Ghoneimy; Ghassan Dbaibo; Rima Wakim; Chiara Azzari; Paolo Palma; Caterina Cancrini; Kelly Capuder; Antonio Condino-Neto; Beatriz T Costa-Carvalho; Joao Bosco Oliveira; Chaim Roifman; David Buchbinder; Attila Kumanovics; Jose Luis Franco; Tim Niehues; Catharina Schuetz; Taco Kuijpers; Christina Yee; Janet Chou; Michel J Masaad; Raif Geha; Gulbu Uzel; Rebecca Gelman; Steven M Holland; Mike Recher; Paul J Utz; Sarah K Browne; Luigi D Notarangelo
Journal:  J Clin Invest       Date:  2015-10-12       Impact factor: 14.808

Review 6.  RAG Deficiency: Two Genes, Many Diseases.

Authors:  Ottavia M Delmonte; Catharina Schuetz; Luigi D Notarangelo
Journal:  J Clin Immunol       Date:  2018-07-25       Impact factor: 8.317

7.  Clinical Manifestations, Mutational Analysis, and Immunological Phenotype in Patients with RAG1/2 Mutations: First Cases Series from Mexico and Description of Two Novel Mutations.

Authors:  Saul Oswaldo Lugo-Reyes; Nina Pastor; Edith González-Serrano; Marco Antonio Yamazaki-Nakashimada; Selma Scheffler-Mendoza; Laura Berron-Ruiz; Guillermo Wakida; Maria Enriqueta Nuñez-Nuñez; Ana Paola Macias-Robles; Aide Tamara Staines-Boone; Edna Venegas-Montoya; Carmen Alaez-Verson; Carolina Molina-Garay; Luis Leonardo Flores-Lagunes; Karol Carrillo-Sanchez; Julie Niemela; Sergio D Rosenzweig; Paul Gaytan; Jorge A Yañez; Ivan Martinez-Duncker; Luigi D Notarangelo; Sara Espinosa-Padilla; Mario Ernesto Cruz-Munoz
Journal:  J Clin Immunol       Date:  2021-05-05       Impact factor: 8.317

Review 8.  Recent advances in understanding RAG deficiencies.

Authors:  Andrew Gennery
Journal:  F1000Res       Date:  2019-02-04

9.  Combined immunodeficiencies: twenty years experience from a single center in Turkey.

Authors:  H Haluk Akar; Turkan Patiroglu; Michael Hershfield; Mirjam van der Burg
Journal:  Cent Eur J Immunol       Date:  2016-01-20       Impact factor: 2.085

10.  A rare case of acquired immunodeficiency associated with myelodysplastic syndrome.

Authors:  Juanjuan Li; Junhui Li; Jianguo Li; Hailan Yao; Fang Liu; James F Gusella; Xiaodong Shi; Xiaoli Chen
Journal:  Mol Genet Genomic Med       Date:  2019-09-10       Impact factor: 2.183

  10 in total

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