Department of Pediatric Surgery, Graduate School of Medicine, Chiba University, Chiba, Japan.
Biomedical Research Center, Chiba University, Chiba, Japan.
Sci Rep. 2021 Feb 4;11(1):3191. doi: 10.1038/s41598-021-82785-1.
The Kif26a protein-coding gene has been identified as a negative regulator of the GDNF-Ret signaling pathway in enteric neurons. The aim of this study was to investigate the influence of genetic background on the phenotype of Kif26a-deficient (KO, -/-) mice. KO mice with both C57BL/6 and BALB/c genetic backgrounds were established. Survival rates and megacolon development were compared between these two strains of KO mice. Functional bowel assessments and enteric neuron histopathology were performed in the deficient mice. KO mice with the BALB/c genetic background survived more than 400 days without evidence of megacolon, while all C57BL/6 KO mice developed megacolon and died within 30 days. Local enteric neuron hyperplasia in the colon and functional bowel abnormalities were observed in BALB/c KO mice. These results indicated that megacolon and enteric neuron hyperplasia in KO mice are influenced by the genetic background. BALB/c KO mice may represent a viable model for functional gastrointestinal diseases such as chronic constipation, facilitating studies on the underlying mechanisms and providing a foundation for the development of treatments.
Kif26a 蛋白编码基因已被确定为肠神经元中 GDNF-Ret 信号通路的负调节剂。本研究旨在探讨遗传背景对 Kif26a 缺陷(KO,-/-)小鼠表型的影响。建立了具有 C57BL/6 和 BALB/c 遗传背景的 KO 小鼠。比较了这两种 KO 小鼠的存活率和巨结肠发育情况。对缺陷小鼠进行了功能性肠评估和肠神经元组织病理学检查。具有 BALB/c 遗传背景的 KO 小鼠存活超过 400 天,没有巨结肠的证据,而所有 C57BL/6 KO 小鼠均发生巨结肠并在 30 天内死亡。在 BALB/c KO 小鼠的结肠中观察到局部肠神经元增生和功能性肠异常。这些结果表明,KO 小鼠的巨结肠和肠神经元增生受遗传背景影响。BALB/c KO 小鼠可能代表一种可行的功能性胃肠疾病模型,如慢性便秘,为研究潜在机制提供了基础,并为治疗方法的开发奠定了基础。