RT Journal Article SR Electronic T1 microCT-Based Skeletal Phenomics in Zebrafish Reveals Virtues of Deep Phenotyping at the Whole-Organism Scale JF bioRxiv FD Cold Spring Harbor Laboratory SP 105569 DO 10.1101/105569 A1 Matthew Hur A1 Charlotte A. Gistelinck A1 Philippe Huber A1 Jane Lee A1 Marjorie H. Thompson A1 Adrian T. Monstad-Rios A1 Claire J. Watson A1 Sarah K. McMenamin A1 Andy Willaert A1 David M Parichy A1 Paul Coucke A1 Ronald Y. Kwon YR 2017 UL http://biorxiv.org/content/early/2017/02/14/105569.abstract AB Phenomics—in-depth phenotyping at the whole-organism scale—holds promise to enhance our fundamental understanding of genes and genomic variation, yet methods in vertebrates are limited. Here, we demonstrate rapid whole-body profiling of hundreds of traits in the axial skeleton of adult zebrafish. We show the potential for vertebral patterns to confer heightened sensitivity, with similar specificity, in discriminating mutant populations compared to analyzing individual vertebrae in isolation, even when the latter is performed at higher resolution. We identify phenotypes associated with human brittle bone disease and thyroid stimulating hormone receptor hyperactivity. Finally, we develop allometric models and show their potential to discriminate mutant phenotypes masked by growth alterations in growth. Our studies demonstrate virtues of whole-body phenomic pattern analysis in a single organ system. The high sensitivity may increase productivity in genetic screens, and facilitate the study genetic variants of smaller effect size, such as those that underlie complex diseases.