Development: For cloche the Bell Tolls

Marlies P Rossmann¹, Yi Zhou¹, Leonard I Zon²

Affiliations

¹ Stem Cell Program and Division of Hematology/Oncology, Children's Hospital Boston, Howard Hughes Medical Institute and Harvard Medical School, Boston, MA 02115, USA. Department of Medical Oncology and Department of Pediatric Oncology, Dana-Farber Cancer Institute, Harvard Medical School, Boston, MA 02215, USA; Department of Stem Cell and Regenerative Biology and Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
² Stem Cell Program and Division of Hematology/Oncology, Children's Hospital Boston, Howard Hughes Medical Institute and Harvard Medical School, Boston, MA 02115, USA. Department of Medical Oncology and Department of Pediatric Oncology, Dana-Farber Cancer Institute, Harvard Medical School, Boston, MA 02215, USA; Department of Stem Cell and Regenerative Biology and Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA. Electronic address: zon@enders.tch.harvard.edu.

PMID: 27728792
PMCID: PMC6126534
DOI: 10.1016/j.cub.2016.08.009

Comment

Development: For cloche the Bell Tolls

Marlies P Rossmann et al. Curr Biol. 2016.

. 2016 Oct 10;26(19):R890-R892.

doi: 10.1016/j.cub.2016.08.009. Epub 2016 Oct 10.

Authors

Marlies P Rossmann¹, Yi Zhou¹, Leonard I Zon²

Affiliations

¹ Stem Cell Program and Division of Hematology/Oncology, Children's Hospital Boston, Howard Hughes Medical Institute and Harvard Medical School, Boston, MA 02115, USA. Department of Medical Oncology and Department of Pediatric Oncology, Dana-Farber Cancer Institute, Harvard Medical School, Boston, MA 02215, USA; Department of Stem Cell and Regenerative Biology and Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA.
² Stem Cell Program and Division of Hematology/Oncology, Children's Hospital Boston, Howard Hughes Medical Institute and Harvard Medical School, Boston, MA 02115, USA. Department of Medical Oncology and Department of Pediatric Oncology, Dana-Farber Cancer Institute, Harvard Medical School, Boston, MA 02215, USA; Department of Stem Cell and Regenerative Biology and Harvard Stem Cell Institute, Harvard University, Cambridge, MA 02138, USA. Electronic address: zon@enders.tch.harvard.edu.

PMID: 27728792
PMCID: PMC6126534
DOI: 10.1016/j.cub.2016.08.009

Abstract

A recent publication identifies npas4l as the gene defective in the well-known cloche mutant that lacks most endothelial as well as hematopoietic cells. This work poses intriguing questions as to the genetic and molecular nature of the origin of hemato-vascular lineages during early embryogenesis.

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Figures

**Figure 1.. Three models describing the generation of the first blood and endothelial cells during embryogenesis and possible location of *npas4l* expression.**
(A) Bipotential hemangioblast giving rise to primitive erythroid and endothelial cells. Adapted from [20]. (B) Endothelial progenitors subspecialize to form hemogenic endothelium as the source of primitive erythroid cells. Adapted from [13]. (C) Endothelial and erythroid progenitors differentiate separately, but the erythroid progenitors require signals from the endothelial ‘microenvironment’. Adapted from [19].

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Comment on

Cloche is a bHLH-PAS transcription factor that drives haemato-vascular specification.
Reischauer S, Stone OA, Villasenor A, Chi N, Jin SW, Martin M, Lee MT, Fukuda N, Marass M, Witty A, Fiddes I, Kuo T, Chung WS, Salek S, Lerrigo R, Alsiö J, Luo S, Tworus D, Augustine SM, Mucenieks S, Nystedt B, Giraldez AJ, Schroth GP, Andersson O, Stainier DY. Reischauer S, et al. Nature. 2016 Jul 14;535(7611):294-8. doi: 10.1038/nature18614. Nature. 2016. PMID: 27411634

References

1. Gritz E, and Hirschi KK (2016). Specification and function of hemogenic endothelium during embryogenesis. Cell Mol. Life Sci 73, 1547–1567. - PMC - PubMed
1. Stainier DY, Weinstein BM, Detrich HW 3rd, Zon LI, and Fishman MC (1995). Cloche, an early acting zebrafish gene, is required by both the endothelial and hematopoietic lineages. Development 121, 3141–3150. - PubMed
1. Reischauer S, Stone OA, Villasenor A, Chi N, Jin SW, Martin M, Lee MT, Fukuda N, Marass M, Witty A, et al. (2016). Cloche is a bHLH-PAS transcription factor that drives haemato-vascular specification. Nature 535, 294–298. - PubMed
1. Liao W, Ho CY, Yan YL, Postlethwait J, and Stainier DY (2000). Hhex and scl function in parallel to regulate early endothelial and blood differentiation in zebrafish. Development 127, 4303–4313. - PubMed
1. Lin Y, Bloodgood BL, Hauser JL, Lapan AD, Koon AC, Kim TK, Hu LS, Malik AN, and Greenberg ME (2008). Activity-dependent regulation of inhibitory synapse development by Npas4. Nature 455, 1198–1204. - PMC - PubMed

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Development: For cloche the Bell Tolls

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Development: For cloche the Bell Tolls

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