Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension

Hiroki Morita; Sabine Mazerbourg; Donna M. Bouley; Ching-Wei Luo; Kazuhiro Kawamura; Yoshimitsu Kuwabara; Helene Baribault; Hui Tian; Aaron J. W. Hsueh

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Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension

机译：LGR5空小鼠的新生儿致死率与强直觉和胃肠道扩张有关。

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The physiological role of an orphan G protein-coupled receptor, LGR5, was investigated by targeted deletion of this seven-transmembrane protein containing a large N-terminal extracellular domain with leucine-rich repeats. LGR5 null mice exhibited 100% neonatal lethality characterized by gastrointestinal tract dilation with air and an absence of milk in the stomach. Gross and histological examination revealed fusion of the tongue to the floor of oral cavity in the mutant newborns and immunostaining of LGR5 expression in the epithelium of the tongue and in the mandible of the wild-type embryos. The observed ankyloglossia phenotype provides a model for understanding the genetic basis of this craniofacial defect in humans and an opportunity to elucidate the physiological role of the LGR5 signaling system during embryonic development.

机译：孤儿G蛋白偶联受体LGR5的生理作用是通过有针对性地删除含有大量N末端胞外域的富含亮氨酸重复序列的七跨膜蛋白来进行的。 LGR5无效的小鼠表现出100％的新生儿致死性，其特征是空气对胃肠道的扩张以及胃中没有牛奶。大体和组织学检查显示，突变体新生儿的舌头融合到口腔底部，舌上皮和野生型下颌骨中的LGR5表达免疫染色。观察到的强直觉表型为理解人类这种颅面缺陷的遗传基础提供了一个模型，并为阐明LGR5信号系统在胚胎发育过程中的生理作用提供了机会。

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《Molecular and Cellular Biology》 |2004年第22期|共8页
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Hiroki Morita; Sabine Mazerbourg; Donna M. Bouley; Ching-Wei Luo; Kazuhiro Kawamura; Yoshimitsu Kuwabara; Helene Baribault; Hui Tian; Aaron J. W. Hsueh;
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中图分类细胞生物学;
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1. Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension [J] . Hiroki Morita, Sabine Mazerbourg, Donna M. Bouley, Molecular and Cellular Biology . 2004,第22期

机译：LGR5空小鼠的新生儿致死率与强直觉和胃肠道扩张有关。
2. Neonatal lethality of neural crest cell-specific Rest knockout mice is associated with gastrointestinal distension caused by aberrations of myenteric plexus [J] . Aoki H, Hara A, Oomori Y, Genes to cells : . 2014,第10期

机译：神经rest细胞特异的Rest基因敲除小鼠的新生儿致死性与肌间神经丛畸变引起的胃肠扩张有关
3. Transgenic rescue of Atg5-null mice from neonatal lethality with neuron-specific expression of ATG5: Systemic analysis of adult Atg5-deficient mice [J] . Yoshii Saori R., Kuma Akiko, Mizushima Noboru Autophagy . 2017,第4期

机译：来自新生儿致死性的ATG5核小鼠的转基因拯救与ATG5的神经元特异性表达：成人ATG5缺陷小鼠的全身分析
4. CURRENT TOPICS IN NEONATAL GASTROINTESTINAL SONOGRAPHY [C] . Cristobal Navas de Solis LV, Jonathan E Palmer Conference of the American College of Veterinary Internal Medicine . 2015

机译：新生儿胃肠道超声检查的当前主题
5. A transgenic mouse that carries the human Fragile-X (FMR1) gene: Neonatal lethality, Fragile-X protein expression and GABA-A receptor beta-subunit expression. [D] . Scalia, Jason. 2003

机译：携带人类脆弱X（FMR1）基因的转基因小鼠：新生儿致死力，脆弱X蛋白表达和GABA-A受体β亚基表达。
6. Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension [O] . Hiroki Morita, Sabine Mazerbourg, Donna M. Bouley, 2004

机译：LGR5空小鼠的新生儿致死率与强直觉和胃肠道扩张有关。
7. Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension [O] . Morita, Hiroki, Mazerbourg, Sabine, Bouley, Donna M., 2004

机译：LGR5空小鼠的新生儿致死率与强直觉和胃肠道扩张有关。
8. Disruption of NBS1 gene leads to early embryonic lethality in homozygous null mice and induces specific cancer in heterozygous mice [R] . 2002

机译：NBs1基因的破坏导致纯合无效小鼠的早期胚胎致死率并诱导杂合小鼠中的特定癌症

Neonatal Lethality of LGR5 Null Mice Is Associated with Ankyloglossia and Gastrointestinal Distension

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