Potential role for the tumor suppressor CYLD in brain and notochord development.

Background: The cylindromatosis (CYLD) tumor suppressor is a microtubule-associated deubiquitinase that plays a critical role in the regulation of cell signaling and contributes to a variety of physiological and pathological processes. However, the functions of CYLD in zebrafish are less well known, particularly with regard to their development and physiology. In this context, we investigated the loss of function of CYLD in zebrafish via transcription activator-like effector nuclease (TALEN)-based gene deletion.

Methods: Semi-quantitative RT-PCR was used to quantify CYLD mRNA expression in zebrafish embryos at various developmental stages. We also performed whole-mount in situ hybridization to further assess the dynamic expression and distribution of CYLD in the entire zebrafish embryos at different stages. In addition, we deleted CYLD in zebrafish with TALENs to investigate its potential impact on embryonic development.

Results: The expression of CYLD mRNA varied during early embryonic development. The CYLD mRNA localized to the brain and notochord of developing zebrafish embryos. Homozygous deletion of CYLD resulted in embryonic death before 8 h post-fertilization.

Conclusions: CYLD appears to play an important role in central nervous system development in zebrafish. Although severe embryonic death restricted analysis of homozygous mutants, further research into the role of CYLD in central nervous system development is warranted.