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Background  Birt-Hogg-Dubé syndrome (BHD) is a dominantly inherited familial cancer syndrome characterised by the development of benign skin fibrofolliculomas, multiple lung and kidney cysts, spontaneous pneumothorax and susceptibility to renal cell carcinoma. BHD is caused by mutations in the gene encoding Folliculin (FLCN). Little is known about what FLCN does in a healthy individual and how best to treat those with BHD. As a first approach to developing a vertebrate model for BHD we aimed to identify the temporal and spatial expression of  flcn  transcripts in the developing zebrafish embryo. To gain insights into the function of  flcn  in a whole organism system we generated a loss of function model of  flcn  by the use of morpholino knockdown in zebrafish.    Results   flcn  is expressed broadly and upregulated in the fin bud, somites, eye and proliferative regions of the brain of the Long-pec stage zebrafish embryos. Together with knockdown phenotypes, expression analysis suggest involvement of  flcn  in zebrafish embryonic brain development. We have utilised the zFucci system, an  in vivo,  whole organism cell cycle assay to study the potential role of  flcn  in brain development. We found that at the 18 somite stage there was a significant drop in cells in the S-M phase of the cell cycle in  flcn  morpholino injected embryos with a corresponding increase of cells in the G1 phase. This was particularly evident in the brain, retina and somites of the embryo. Timelapse analysis of the head region of  flcn  morpholino injected and mismatch control embryos shows the temporal dynamics of cell cycle misregulation during development.    Conclusions  In conclusion we show that zebrafish  flcn  is expressed in a non-uniform manner and is likely required for the maintenance of correct cell cycle regulation during embryonic development. We demonstrate the utilisation of the zFucci system in testing the role of  flcn  in cell proliferation and suggest a function for  flcn  in regulating cell proliferation in vertebrate embryonic brain development.    Electronic supplementary material  The online version of this article (doi:10.1186/s12861-016-0119-8) contains supplementary material, which is available to authorized users.

Expression and knockdown of zebrafish folliculin suggests requirement for embryonic brain morphogenesis