TY - JOUR T1 - microCT-Based Skeletal Phenomics in Zebrafish Reveals Virtues of Deep Phenotyping at the Whole-Organism Scale JF - bioRxiv DO - 10.1101/105569 SP - 105569 AU - Matthew Hur AU - Charlotte A. Gistelinck AU - Philippe Huber AU - Jane Lee AU - Marjorie H. Thompson AU - Adrian T. Monstad-Rios AU - Claire J. Watson AU - Sarah K. McMenamin AU - Andy Willaert AU - David M Parichy AU - Paul Coucke AU - Ronald Y. Kwon Y1 - 2017/01/01 UR - http://biorxiv.org/content/early/2017/03/02/105569.abstract N2 - Phenomics–in-depth phenotyping at the whole-organism scale-holds promise to significantly enhance our functional 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 deep phenotyping in a single organ system. Phenomic pattern analysis may increase productivity in genetic screens, and facilitate the study genetic variants of smaller effect size, such as those that underlie complex diseases. ER -