To elucidate molecular mechanisms contributing to the disease phenotype in col10a1 medaka (Oryzias latipes) mutants, we isolated col10a1:nlGFP cells from heterozygous col10a1 medaka mutants and wild-type siblings via fluorescence activated cell sorting (FACS) and performed RNA-seq. Comparison of the transcriptome profiles indicated elevated ER stress, dysregulated cell polarity and reduced ECM production in heterozygous col10a1 mutants. Overall design: Comparative gene expression profiling analysis of RNA-seq data for col10a1:nlGFP-expressing cells in col10a1 medaka mutants and wild-type siblings. Two biological replicates were analyzed for heterozygous col10a1 mutants and three biological replicates were analyzed for wild-type siblings.