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Development 128, 3975-3986 (2001)
© 2001 The Company of Biologists Limited

Severe nasal clefting and abnormal embryonic apoptosis in Alx3/Alx4 double mutant mice

Annemiek Beverdam, Antje Brouwer, Mark Reijnen, Jeroen Korving and Frits Meijlink*

Hubrecht Laboratory, Netherlands Institute for Developmental Biology, PO Box 85164, 3508AD Utrecht, The Netherlands

*Author for correspondence (e-mail: frits{at}niob.knaw.nl)

Accepted July 18, 2001

A group of mouse aristaless-related genes has been implicated in functions in the development of the craniofacial skeleton. We have generated an Alx3 mutant allele in which the lacZ coding sequence is inserted in-frame in the Alx3 gene and the sequences encoding the conserved protein domains are deleted. Mice homozygous for this null allele are indistinguishable from wild-type mice. Compound mutants of Alx3 and Alx4, however, show severe craniofacial abnormalities that are absent in Alx4 single mutants. Alx3/Alx4 double mutant newborn mice have cleft nasal regions. Most facial bones and many other neural crest derived skull elements are malformed, truncated or even absent. The craniofacial defects in Alx3/Alx4 double mutant embryos become anatomically manifest around embryonic day 10.5, when the nasal processes appear to be abnormally positioned. This most probably leads to a failure of the medial nasal processes to fuse in the facial midline and subsequently to the split face phenotype. We detected a significant increase in apoptosis localised in the outgrowing frontonasal process in embryonic day 10.0 double mutant embryos, which we propose to be the underlying cause of the subsequent malformations.

Key words: Cleft face, Homeobox gene, Craniofacial development, Apoptosis, Mouse, Aristaless-related genes, Cephalic neural crest, Strong’s luxoid, Limb development




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© The Company of Biologists Ltd 2001