son is necessary for proper vertebrate blood development.
son is necessary for proper vertebrate blood development.
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DOI：
10.1371/journal.pone.0247489
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发表时间：
2021
期刊：
影响因子：
3.7
通讯作者：
Stachura DL
中科院分区：
文献类型：
--
作者：
Belmonte RL;Engbretson IL;Kim JH;Cajias I;Ahn EE;Stachura DL
SON基因位于人类21号染色体（21q22.11）上，被认为与伴随唐氏综合征的造血系统疾病有关。此外，SON是一种RNA剪接因子，在白血病相关基因的转录中发挥作用。以前，我们发现SON突变会导致人类和斑马鱼在胚胎发育早期的脊柱和大脑畸形。为了研究SON在正常造血中的作用，我们用特定的吗啉代在单细胞发育阶段减少了斑马鱼SON的斑马鱼同源物的表达。除了大脑和脊髓畸形，我们还观察到在儿子击倒后异常的血细胞水平。然后，我们研究了当son水平降低时，血液生产如何改变。当用lcr：GFP转基因鱼可视化时，son水平降低导致红细胞量降低。当检查mpx：GFP鱼时，用cd41：GFP鱼也观察到血小板减少，并且骨髓细胞减少。我们还观察到T细胞数量显着减少，用lck：GFP鱼可见。然而，当我们检查他们的造血干细胞和祖细胞（HSPC）时，我们没有看到集落形成能力的差异。这些研究表明，儿子是先天性和适应性免疫系统的适当分化必不可少的，并进一步研究确定血液发育过程中涉及的分子途径应阐明有关脊椎动物HSPC的产生，增殖和分化的重要信息。
The gene SON is on human chromosome 21 (21q22.11) and is thought to be associated with hematopoietic disorders that accompany Down syndrome. Additionally, SON is an RNA splicing factor that plays a role in the transcription of leukemia-associated genes. Previously, we showed that mutations in SON cause malformations in human and zebrafish spines and brains during early embryonic development. To examine the role of SON in normal hematopoiesis, we reduced expression of the zebrafish homolog of SON in zebrafish at the single-cell developmental stage with specific morpholinos. In addition to the brain and spinal malformations we also observed abnormal blood cell levels upon son knockdown. We then investigated how blood production was altered when levels of son were reduced. Decreased levels of son resulted in lower amounts of red blood cells when visualized with lcr:GFP transgenic fish. There were also reduced thrombocytes seen with cd41:GFP fish, and myeloid cells when mpx:GFP fish were examined. We also observed a significant decrease in the quantity of T cells, visualized with lck:GFP fish. However, when we examined their hematopoietic stem and progenitor cells (HSPCs), we saw no difference in colony-forming capability. These studies indicate that son is essential for the proper differentiation of the innate and adaptive immune system, and further investigation determining the molecular pathways involved during blood development should elucidate important information about vertebrate HSPC generation, proliferation, and differentiation.
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影响因子：
16
作者：
Kim JH;Baddoo MC;Park EY;Stone JK;Park H;Butler TW;Huang G;Yan X;Pauli-Behn F;Myers RM;Tan M;Flemington EK;Lim ST;Ahn EY
通讯作者：
Ahn EY
影响因子：
21.3
作者：
Damm EW;Clements WK
通讯作者：
Clements WK
影响因子：
2.7
作者：
Ganis JJ;Hsia N;Trompouki E;de Jong JL;DiBiase A;Lambert JS;Jia Z;Sabo PJ;Weaver M;Sandstrom R;Stamatoyannopoulos JA;Zhou Y;Zon LI
通讯作者：
Zon LI
影响因子：
20.3
作者：
Lin, HF;Traver, D;Handin, RI
通讯作者：
Handin, RI
影响因子：
1.2
作者：
Berrun, A. C.;Stachura, D. L.
通讯作者：
Stachura, D. L.