Forward Genetic Screens in Zebrafish Identify Pre-mRNA-Processing Pathways Regulating Early T Cell Development

Norimasa Iwanami, Katarzyna Sikora, Andreas S. Richter, Maren Mönnich, Lucia Guerri, Cristian Soza-Ried, Divine Fondzenyuy Lawir, Fernando Mateos, Isabell Hess, Connor P. O'Meara, Michael Schorpp, Thomas Boehm

Resultado de la investigación: Contribución a una revistaArtículo

8 Citas (Scopus)

Resumen

Lymphocytes represent basic components of vertebrate adaptive immune systems, suggesting the utility of non-mammalian models to define the molecular basis of their development and differentiation. Our forward genetic screens in zebrafish for recessive mutations affecting early T cell development revealed several major genetic pathways. The identification of lineage-specific transcription factors and specific components of cytokine signaling and DNA replication and/or repair pathways known from studies of immunocompromised mammals provided an evolutionary cross-validation of the screen design. Unexpectedly, however, genes encoding proteins required for pre-mRNA processing were enriched in the collection of mutants identified here. In both zebrafish and mice, deficiency of the splice regulator TNPO3 impairs intrathymic T cell differentiation, illustrating the evolutionarily conserved and cell-type-specific functions of certain pre-mRNA-processing factors for T cell development.

Idioma originalInglés
Páginas (desde-hasta)2259-2270
Número de páginas12
PublicaciónCell Reports
Volumen17
N.º9
DOI
EstadoPublicada - 22 nov 2016
Publicado de forma externa

Áreas temáticas de ASJC Scopus

  • Bioquímica, genética y biología molecular (todo)

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    Iwanami, N., Sikora, K., Richter, A. S., Mönnich, M., Guerri, L., Soza-Ried, C., Lawir, D. F., Mateos, F., Hess, I., O'Meara, C. P., Schorpp, M., & Boehm, T. (2016). Forward Genetic Screens in Zebrafish Identify Pre-mRNA-Processing Pathways Regulating Early T Cell Development. Cell Reports, 17(9), 2259-2270. https://doi.org/10.1016/j.celrep.2016.11.003