Literature DB >> 19011808

Omenn syndrome with mutation in RAG1 gene.

I Cherkaoui Jaouad1, K Ouldim, S Ali Ou Alla, Y Kriouile, A Villa, A Sefiani.   

Abstract

Omenn syndrome is a form of severe combined immunodeficiency associated with erythrodermia, hepatosplenomegaly, lymphadenopathy, and alopecia. Inherited hypomorphic mutations in the recombination activating genes 1 and 2 (RAG1 and RAG2) and in ARTEMIS genes and more recently defects in IL7RA, and RMRP genes have been described to be responsible of this peculiar immunodeficiency. The authors report here a Moroccan patient of four-months-old with classical features of Omenn syndrome, carrying a deletion at the N terminal part of RAG1. Early recognition of this condition is important for genetic counseling and early treatment.

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Year:  2008        PMID: 19011808     DOI: 10.1007/s12098-008-0197-0

Source DB:  PubMed          Journal:  Indian J Pediatr        ISSN: 0019-5456            Impact factor:   1.967


  15 in total

1.  Mutations in the RNA component of RNase mitochondrial RNA processing might cause Omenn syndrome.

Authors:  Chaim M Roifman; Yiping Gu; Amos Cohen
Journal:  J Allergy Clin Immunol       Date:  2006-04       Impact factor: 10.793

2.  Oligoclonal expansion of CD45RO+ T lymphocytes in Omenn syndrome.

Authors:  T O Harville; D M Adams; T A Howard; R E Ware
Journal:  J Clin Immunol       Date:  1997-07       Impact factor: 8.317

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.  N-terminal truncated human RAG1 proteins can direct T-cell receptor but not immunoglobulin gene rearrangements.

Authors:  J G Noordzij; N S Verkaik; N G Hartwig; R de Groot; D C van Gent; J J van Dongen
Journal:  Blood       Date:  2000-07-01       Impact factor: 22.113

Review 5.  Omenn syndrome: a lack of tolerance on the background of deficient lymphocyte development and maturation.

Authors:  Manfred Hönig; Klaus Schwarz
Journal:  Curr Opin Rheumatol       Date:  2006-07       Impact factor: 5.006

6.  Artemis, a novel DNA double-strand break repair/V(D)J recombination protein, is mutated in human severe combined immune deficiency.

Authors:  D Moshous; I Callebaut; R de Chasseval; B Corneo; M Cavazzana-Calvo; F Le Deist; I Tezcan; O Sanal; Y Bertrand; N Philippe; A Fischer; J P de Villartay
Journal:  Cell       Date:  2001-04-20       Impact factor: 41.582

7.  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

8.  Differentiation of materno-fetal GVHD from Omenn's syndrome in pre-BMT patients with severe combined immunodeficiency.

Authors:  A L Appleton; A Curtis; J Wilkes; A J Cant
Journal:  Bone Marrow Transplant       Date:  1994-07       Impact factor: 5.483

9.  Omenn syndrome in an infant with IL7RA gene mutation.

Authors:  Silvia Giliani; Carmen Bonfim; Genevieve de Saint Basile; Gaetana Lanzi; Nicole Brousse; Adriana Koliski; Mariester Malvezzi; Alain Fischer; Luigi D Notarangelo; Francoise Le Deist
Journal:  J Pediatr       Date:  2006-02       Impact factor: 4.406

10.  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

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  2 in total

1.  Hypomorphic Rag mutations can cause destructive midline granulomatous disease.

Authors:  Suk See De Ravin; Edward W Cowen; Kol A Zarember; Narda L Whiting-Theobald; Douglas B Kuhns; Netanya G Sandler; Daniel C Douek; Stefania Pittaluga; Pietro L Poliani; Yu Nee Lee; Luigi D Notarangelo; Lei Wang; Frederick W Alt; Elizabeth M Kang; Joshua D Milner; Julie E Niemela; Mary Fontana-Penn; Sara H Sinal; Harry L Malech
Journal:  Blood       Date:  2010-05-20       Impact factor: 22.113

Review 2.  Proposal for a 6-step approach for differential diagnosis of neonatal erythroderma.

Authors:  E Cuperus; A Bygum; L Boeckmann; C Bodemer; M C Bolling; M Caproni; A Diociaiuti; S Emmert; J Fischer; A Gostynski; S Guez; M E van Gijn; K Hannulla-Jouppi; C Has; A Hernández-Martín; A E Martinez; J Mazereeuw-Hautier; M Medvecz; I Neri; V Sigurdsson; K Suessmuth; H Traupe; V Oji; S G M A Pasmans
Journal:  J Eur Acad Dermatol Venereol       Date:  2022-03-15       Impact factor: 9.228

  2 in total

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