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Human Molecular Genetics Advance Access originally published online on July 24, 2008
Human Molecular Genetics 2008 17(20):3204-3211; doi:10.1093/hmg/ddn216
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© The Author 2008. Published by Oxford University Press. All rights reserved. For Permissions, please email: journals.permissions@oxfordjournals.org

Deficiency of ribosomal protein S19 during early embryogenesis leads to reduction of erythrocytes in a zebrafish model of Diamond-Blackfan anemia

Tamayo Uechi, Yukari Nakajima, Anirban Chakraborty, Hidetsugu Torihara, Sayomi Higa and Naoya Kenmochi*

Frontier Science Research Center, University of Miyazaki, Miyazaki 889-1692, Japan

* To whom correspondence should be addressed at: Frontier Science Research Center, University of Miyazaki, 5200 Kihara, Kiyotake, Miyazaki 889-1692, Japan. Tel: +81 985859084; Fax: +81 985859084; Email: kenmochi{at}med.miyazaki-u.ac.jp or t_uechi{at}med.miyazaki-u.ac.jp

Received May 2, 2008; Accepted July 18, 2008

Ribosomes are responsible for protein synthesis in all cells. Ribosomal protein S19 (RPS19) is one of the 79 ribosomal proteins (RPs) in vertebrates. Heterozygous mutations in RPS19 have been identified in 25% of patients with Diamond-Blackfan anemia (DBA), but the relationship between RPS19 mutations and the pure red-cell aplasia of DBA is unclear. In this study, we developed an RPS19-deficient zebrafish by knocking down rps19 using a Morpholino antisense oligo. The RPS19-deficient animals showed a dramatic decrease in blood cells as well as deformities in the head and tail regions at early developmental stages. These phenotypes were rescued by injection of zebrafish rps19 mRNA, but not by injection of rps19 mRNAs with mutations that have been identified in DBA patients. Our results indicate that rps19 is essential for hematopoietic differentiation during early embryogenesis. The effects were specific to rps19, but knocking down the genes for three other RPs, rpl35, rpl35a and rplp2, produced similar phenotypes, suggesting that these genes might have a common function in zebrafish erythropoiesis. The RPS19-deficient zebrafish will provide a valuable tool for investigating the molecular mechanisms of DBA development in humans.


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