Nature immunology · 2019
Syndromic immune disorder caused by a viable hypomorphic allele of spliceosome component Snrnp40.
СтатьяПоддержка NIH: внешняя программаПоддержка исследования: вне правительства СШАНазвание и аннотация: Перевод в очереди
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Zhang Duanwu, Yue Tao, Choi Jin Huk, Nair-Gill Evan, Zhong Xue, Wang Kuan-Wen, Zhan Xiaoming, Li Xiaohong, Choi Mihwa, Tang Miao, Quan Jiexia, Hildebrand Sara, Moresco Eva Marie Y, Beutler Bruce
Аннотация
We report a new immunodeficiency disorder in mice caused by a viable hypomorphic mutation of Snrnp40, an essential gene encoding a subunit of the U5 small nuclear ribonucleoprotein (snRNP) complex of the spliceosome. Snrnp40 is ubiquitous but strongly expressed in lymphoid tissue. Homozygous mutant mice showed hypersusceptibility to infection by murine cytomegalovirus and multiple defects of lymphoid development, stability and function. Cell-intrinsic defects of hematopoietic stem cell differentiation also affected homozygous mutants. SNRNP40 deficiency in primary hematopoietic stem cells or T cells or the EL4 cell line increased the frequency of splicing errors, mostly intron retention, in several hundred messenger RNAs. Altered expression of proteins associated with immune cell function was also observed in Snrnp40-mutant cells. The immunological consequences of SNRNP40 deficiency presumably result from cumulative, moderate effects on processing of many different mRNA molecules and secondary reductions in the expression of critical immune proteins, yielding a syndromic immune disorder.
Предметные рубрики MeSH
AllelesAnimalsCell LineClustered Regularly Interspaced Short Palindromic RepeatsDisease SusceptibilityHematopoietic Stem CellsHerpesviridae InfectionsImmunologic Deficiency SyndromesLymphopoiesisMiceMice, Inbred C57BLMice, KnockoutMuromegalovirusMutationRNA SplicingRibonucleoprotein, U5 Small NuclearSpliceosomesT-Lymphocytes