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Related Concept Videos

Neurulation01:30

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Neurulation is the embryological process which forms the precursors of the central nervous system and occurs after gastrulation has established the three primary cell layers of the embryo: ectoderm, mesoderm, and endoderm. In humans, the majority of this system is formed via primary neurulation, in which the central portion of the ectoderm—originally appearing as a flat sheet of cells—folds upwards and inwards, sealing off to form a hollow neural tube. As development proceeds, the anterior...
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Real-time Bioluminescence Imaging of Notch Signaling Dynamics during Murine Neurogenesis
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Published on: December 12, 2019

The transcription factor Nfix is essential for normal brain development.

Christine E Campbell1, Michael Piper, Céline Plachez

  • 1Dept. of Biochemistry and New York State Center of Excellence in Bioinformatics and Life Sciences, State University of New York at Buffalo, 3435 Main St,, Buffalo, NY 14214, USA. cc59@buffalo.edu

BMC Developmental Biology
|May 15, 2008
PubMed
Summary

Nuclear Factor I-X (Nfix) is essential for normal brain development, affecting neural stem cell homeostasis and causing developmental delays. Nfix-deficient mice exhibit unique brain defects and perinatal lethality, independent of nutritional issues.

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Area of Science:

  • Neuroscience
  • Developmental Biology
  • Genetics

Background:

  • The Nuclear Factor I (NFI) family of transcription factors is crucial for organ system development.
  • Previous studies showed varied defects in Nfia, Nfib, and Nfic knockout mice, with Nfix largely uncharacterized.
  • Nfix disruption was previously suggested to cause skeletal and brain development defects.

Purpose of the Study:

  • To investigate the specific role of Nfix in brain development.
  • To characterize the phenotypes of Nfix-deficient mice.

Main Methods:

  • Cre-recombinase mediated excision of the 2nd exon of Nfix to create Nfix knockout mice.
  • Phenotypic analysis of brain morphology, growth rate, and survival.
  • Assessment of developmental delays (eye and ear opening).
  • Nutritional rescue experiments using soft dough chow.

Main Results:

  • Nfix deficiency leads to distinct brain defects, including aberrant Pax6- and doublecortin-positive cells, increased brain weight, cortical expansion, and hippocampal abnormalities.
  • Nfix-/- mice exhibit a decreased growth rate, weight loss, and perinatal lethality around P22.
  • Delayed eye and ear canal opening suggest impaired epithelial development.
  • Nutritional supplementation partially rescues growth but does not eliminate brain defects or lethality.

Conclusions:

  • Nfix is essential for normal brain development and potentially for neural stem cell homeostasis.
  • Brain morphology defects and developmental delays in Nfix-/- mice are independent of nutritional deprivation.
  • Nfix plays a critical role in forebrain development, distinct from other NFI family members.