Mutation in Hairless Dogs Implicates FOXI3 in Ectodermal Development

Mexican and Peruvian hairless dogs and Chinese crested dogs are characterized by missing hair and teeth, a phenotype termed canine ectodermal dysplasia (CED). CED is inherited as a monogenic autosomal semidominant trait. With genomewide association analysis we mapped the CED mutation to a 102-kilo-b...

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Published in:Science (American Association for the Advancement of Science) 2008-09, Vol.321 (5895), p.1462-1462
Main Authors: Drögemüller, Cord, Karlsson, Elinor K, Hytönen, Marjo K, Perloski, Michele, Dolf, Gaudenz, Sainio, Kirsi, Lohi, Hannes, Lindblad-Toh, Kerstin, Leeb, Tosso
Format: Article
Language:English
Subjects:
Amino Acid Sequence
Animals
Brevia
Chromosome Mapping
Dog Diseases - genetics
Dogs
Dogs - genetics
Ectoderm - embryology
Ectoderm - metabolism
Ectodermal Dysplasia - genetics
Ectodermal Dysplasia - veterinary
Ectodysplasins - metabolism
Exons
Female
Forkhead Transcription Factors - chemistry
Forkhead Transcription Factors - genetics
Forkhead Transcription Factors - physiology
Frameshift Mutation
Gene Duplication
Genes
Genetic mutation
Genomes
Genomics
Genotype & phenotype
Hair
Hair - embryology
Hair - metabolism
Haplotypes
Male
Mice
Molecular Sequence Data
Mutant proteins
Mutant Proteins - chemistry
Mutant Proteins - genetics
Mutant Proteins - physiology
Mutation
NATURAL SCIENCES
NATURVETENSKAP
Pedigree
Phenotypes
Polymorphism, Single Nucleotide
Sequence Analysis, DNA
Signal Transduction
Teeth
Tooth - embryology
Tooth - metabolism
Vibrissae - embryology
Vibrissae - metabolism
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Summary:Mexican and Peruvian hairless dogs and Chinese crested dogs are characterized by missing hair and teeth, a phenotype termed canine ectodermal dysplasia (CED). CED is inherited as a monogenic autosomal semidominant trait. With genomewide association analysis we mapped the CED mutation to a 102-kilo-base pair interval on chromosome 17. The associated interval contains a previously uncharacterized member of the forkhead box transcription factor family (FOXI3), which is specifically expressed in developing hair and teeth. Mutation analysis revealed a frameshift mutation within the FOXI3 coding sequence in hairless dogs. Thus, we have identified FOXI3 as a regulator of ectodermal development.
ISSN:0036-8075
1095-9203
1095-9203
DOI:10.1126/science.1162525