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Human Molecular Genetics, 2001, Vol. 10, No. 12 1325-1334
© 2001 Oxford University Press

Neural tube defects and neuroepithelial cell death in Tulp3 knockout mice

Akihiro Ikeda, Sakae Ikeda, Thomas Gridley, Patsy M. Nishina and Jürgen K. Naggert+

The Jackson Laboratory, 600 Main Street, Bar Harbor, ME 04609, USA

The tubby-like protein 3 (Tulp3) gene has been identified as a member of a small novel gene family which is primarily neuronally expressed. Mutations in two of the family members, tub and tulp1, have been shown to cause neurosensory disorders. To determine the in vivo function of Tulp3, we have generated a germline mutation in the mouse Tulp3 gene by homologous recombination. Embryos homozygous for the Tulp3 mutant allele exhibit failure of neural tube closure, and die by embryonic day 14.5. Failure of cranial neural tube closure coincided with increased neuroepithelial apoptosis specifically in the hindbrain and the caudal neural tube. In addition, the number of ßIII-tubulin positive cells is significantly decreased in the hindbrain of Tulp3–/– embryos. These results suggest that disruption of the Tulp3 gene affects the development of a neuronal cell population. Interestingly, some Tulp3 heterozygotes also manifest embryonic lethality with neuroepithelial cell death. Our results demonstrate that the Tulp3 gene is essential for embryonic development in mice.

+ To whom correspondence should be addressed. Tel: +1 207 288 6384; Fax: +1 207 288 6079; Email: jkn@jax.org


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