PT - JOURNAL ARTICLE AU - Harwin Sidik AU - Christy J. Ang AU - Mahmoud A. Pouladi TI - Huntingtin confers fitness but is not embryonically essential in zebrafish development AID - 10.1101/615591 DP - 2019 Jan 01 TA - bioRxiv PG - 615591 4099 - http://biorxiv.org/content/early/2019/04/28/615591.short 4100 - http://biorxiv.org/content/early/2019/04/28/615591.full AB - Attempts to constitutively knockout HTT in rodents resulted in embryonic lethality, curtailing efforts to study HTT function later in development. Here we show that HTT is dispensable for early zebrafish development, contrasting published zebrafish morpholino experiment results. Homozygous HTT knockouts were embryonically viable and appeared developmentally normal through juvenile stages. Comparison of adult fish revealed significant reduction in body size and fitness in knockouts compared to hemizygotes and wildtype fish, indicating an important role for wildtype HTT in postnatal development. Our zebrafish model provides an opportunity to examine the function of wildtype HTT later in development.