Identification of Gene Mutations and Fusion Genes in Patients with Sezary Syndrome


Por: Prasad, A, Rabionet, R, Espinet, B, Zapata, L, Puiggros, A, Melero, C, Puig, A, Sarria-Trujillo, Y, Ossowski, S, Garcia-Muret, MP, Estrach, T, Servitje, O, Lopez-Lerma, I, Gallardo, F, Pujol, RM, Estivill, X

Publicada: 1 jul 2016
Resumen:
Sezary syndrome is a leukemic form of cutaneous T-cell lymphoma with an aggressive clinical course. The genetic etiology of the disease is poorly understood, with chromosomal abnormalities and mutations in some genes being involved in the disease. The goal of our study was to understand the genetic basis of the disease by looking for driver gene mutations and fusion genes in 15 erythrodermic patients with circulating Sezary cells, 14 of them fulfilling the diagnostic criteria of Sezary syndrome. We have discovered genes that could be involved in the pathogenesis of Sezary syndrome. Some of the genes that are affected by somatic point mutations include ITPR1, ITPR2, DSC1, RIPK2, IL6, and RAG2, with some of them mutated in more than one patient. We observed several somatic copy number variations shared between patients, including deletions and duplications of large segments of chromosome 17. Genes with potential function in the T-cell receptor signaling pathway and tumorigenesis were disrupted in Sezary syndrome patients, for example, CBLB, RASA2, BCL7C, RAMP3, TBRG4, and DAD1. Furthermore, we discovered several fusion events of interest involving RASA2, NFKB2, BCR, FASN, ZEB1, TYK2, and SGMS1. Our work has implications for the development of potential therapeutic approaches for this aggressive disease.

Filiaciones:
Prasad, A:
 Barcelona Inst Sci & Technol, Ctr Genom Regulat CRG, Dr Aiguader 88, Barcelona 08003, Spain

 UPF, Barcelona, Spain

 CIBER Epidemiol & Publ Hlth CIBERESP, Barcelona, Spain

 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

Rabionet, R:
 Barcelona Inst Sci & Technol, Ctr Genom Regulat CRG, Dr Aiguader 88, Barcelona 08003, Spain

 UPF, Barcelona, Spain

 CIBER Epidemiol & Publ Hlth CIBERESP, Barcelona, Spain

 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

Espinet, B:
 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

 Hosp del Mar, Lab Mol Cytogenet, Pathol Serv, Barcelona, Spain

Zapata, L:
 Barcelona Inst Sci & Technol, Ctr Genom Regulat CRG, Dr Aiguader 88, Barcelona 08003, Spain

 UPF, Barcelona, Spain

 CIBER Epidemiol & Publ Hlth CIBERESP, Barcelona, Spain

Puiggros, A:
 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

 Hosp del Mar, Lab Mol Cytogenet, Pathol Serv, Barcelona, Spain

Melero, C:
 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

 Hosp del Mar, Lab Mol Cytogenet, Pathol Serv, Barcelona, Spain

Puig, A:
 Barcelona Inst Sci & Technol, Ctr Genom Regulat CRG, Dr Aiguader 88, Barcelona 08003, Spain

 UPF, Barcelona, Spain

 CIBER Epidemiol & Publ Hlth CIBERESP, Barcelona, Spain

 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

Sarria-Trujillo, Y:
 Barcelona Inst Sci & Technol, Ctr Genom Regulat CRG, Dr Aiguader 88, Barcelona 08003, Spain

 UPF, Barcelona, Spain

 CIBER Epidemiol & Publ Hlth CIBERESP, Barcelona, Spain

 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

Ossowski, S:
 Barcelona Inst Sci & Technol, Ctr Genom Regulat CRG, Dr Aiguader 88, Barcelona 08003, Spain

 UPF, Barcelona, Spain

 CIBER Epidemiol & Publ Hlth CIBERESP, Barcelona, Spain

Garcia-Muret, MP:
 Hosp Santa Creu & Sant Pau, Dermatol Serv, Barcelona, Spain

Estrach, T:
 Hosp Clin Barcelona, Dept Dermatol, Barcelona, Spain

Servitje, O:
 Bellvitge Hosp, Dermatol Serv, Barcelona, Spain

Lopez-Lerma, I:
 Hosp Valle De Hebron, Dept Dermatol, Barcelona, Spain

Gallardo, F:
 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

 Hosp del Mar, Dermatol Serv, Barcelona, Spain

Pujol, RM:
 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

 Hosp del Mar, Dermatol Serv, Barcelona, Spain

Estivill, X:
 Barcelona Inst Sci & Technol, Ctr Genom Regulat CRG, Dr Aiguader 88, Barcelona 08003, Spain

 UPF, Barcelona, Spain

 CIBER Epidemiol & Publ Hlth CIBERESP, Barcelona, Spain

 Hosp del Mar Med Res Inst IMIM, Barcelona, Spain

 Sidra Med & Res Ctr, Expt Genet, Doha, Qatar
ISSN: 0022202X





JOURNAL OF INVESTIGATIVE DERMATOLOGY
Editorial
ELSEVIER SCIENCE INC, STE 800, 230 PARK AVE, NEW YORK, NY 10169 USA, Estados Unidos America
Tipo de documento: Article
Volumen: 136 Número: 7
Páginas: 1490-1499
WOS Id: 000379016400026
ID de PubMed: 27039262
imagen Green Published, Hybrid Gold

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