Anosmin-1 is essential for neural crest and cranial placodes formation in Xenopus
Highlights: • Human mutations in ANOS1 cause Kallmann syndrome. • Anos1 is a target of Pax3 and Zic1 in Xenopus laevis. • Anos1 is expressed in cranial neural crest cells and cranial placode derivatives. • Anos1 knockdown affects both neural crest and sensory organs formation. • Anos1-depleted Xenopus is a model to study the pathogenesis of Kallmann syndrome. During embryogenesis vertebrates develop a complex craniofacial skeleton associated with sensory organs. These structures are primarily derived from two embryonic cell populations the neural crest and cranial placodes, respectively. Neural crest cells and cranial placodes are specified through the integrated action of several families of signaling molecules, and the subsequent activation of a complex network of transcription factors. Here we describe the expression and function of Anosmin-1 (Anos1), an extracellular matrix protein, during neural crest and cranial placodes development in Xenopus laevis. Anos1 was identified as a target of Pax3 and Zic1, two transcription factors necessary and sufficient to generate neural crest and cranial placodes. Anos1 is expressed in cranial neural crest progenitors at early neurula stage and in cranial placode derivatives later in development. We show that Anos1 function is required for neural crest and sensory organs development in Xenopus, consistent with the defects observed in Kallmann syndrome patients carrying a mutation in ANOS1. These findings indicate that anos1 has a conserved function in the development of craniofacial structures, and indicate that anos1-depleted Xenopus embryos represent a useful model to analyze the pathogenesis of Kallmann syndrome.