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Congress Abstracts - Society for Developmental Biology

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Program/Abstract # 257<br />

Fritz Governs Ciliogenesis in Xenopus laevis.<br />

Kim, Su Kyoung (Univ of Texas-Austin, USA); Park, Tae Joo (Ulsan Metropolitan City, Korea); Abitua, Phil B. (University of<br />

Cali<strong>for</strong>nia-Berkeley, USA); Walling<strong>for</strong>d, John B. (Univ of Texas-Austin, USA)<br />

Cilia are evolutionarily conserved microtubule-based organelles projecting from nearly all vertebrate cells, and ciliary defects result in<br />

a variety of human disorders known as ciliopathies. Recent studies have shown that several planar cell polarity (PCP) proteins are<br />

essential <strong>for</strong> cilia functions. Here, we focused on Fritz, known as a novel PCP effector protein in Drosophila, in multi-ciliated cells in<br />

the epidermis of Xenopus laevis embryos. To investigate the role of Fritz, using confocal and scanning electron microscopy, we<br />

discovered that Fritz localizes along the ciliary axonemes and that knockdown of Fritz causes severe reductions in axonemes length<br />

and number. Then, using pull-downs and mass-spectrometry, we identified the Chaperonin Containing T-complex polypeptide 1<br />

(CCT) and septin as interacting partners of Fritz. CCT is the key chaperonin interacting with septins, and both have been implicated in<br />

ciliogenesis. Using tagged CCT subunit constructs, we found that the tagged CCTα and CCTε co-localize with Fritz along the ciliary<br />

axonemes of multi-ciliated cells. Knocking-down of Fritz results in the accumulation of CCT at the apical cytoplasm in multi-ciliated<br />

cells; however, we confirmed that Fritz does not affect the CCT holoenzyme assembly. Septins, another interacting partner of Fritz,<br />

are novel cytoskeletal elements. Using septin antibodies, we found that endogenous septins also localize along the axonemes and<br />

accumulate in the apical cytoplasm of multi-ciliated cells in Fritz morphants. Similar ciliary defects were observed in septins<br />

morphants. Our data reveal that Fritz is essential <strong>for</strong> ciliogenesis, and that CCT and septin may interact with Fritz to control<br />

ciliogenesis in Xenopus multi-ciliated cells.<br />

Program/Abstract # 258<br />

Molecular basis of principles of regeneration: distalization and intercalation<br />

Agata, Kiyokazu, (Kyoto University, Japan<br />

Regeneration is always conducted under the control of positional in<strong>for</strong>mation. The distal portion of the body is <strong>for</strong>med immediately<br />

after wound healing around the cut surface (a step called ‘distalization’), and interaction of the newly <strong>for</strong>med distal portion and the<br />

remaining proximal portion may next induce reorganization of positional in<strong>for</strong>mation, and lost tissues are then intercalatively<br />

generated to restore the original structures (‘intercalation’). We proposed that this is probably a general principal of regeneration from<br />

invertebrates to vertebrates (Agata et al., Dev. Growth Differ., 49, 73–78, 2007). Recently we found that the molecular basis<br />

underlying diatalization and intercalation is also very similar among different regeneration events, although the tissues acquiring distal<br />

characteristics and cells participating in the regeneration of lost tissues vary among different animals and different regeneration<br />

systems. Here we will compare the molecular mechanisms of planarian regeneration and newt limb regeneration and then discuss<br />

common aspects of regeneration.<br />

Program/Abstract # 259<br />

GXD: A Gene Expression Resource <strong>for</strong> <strong>Developmental</strong> Biologists<br />

Smith, Constance M.; Finger, Jacqueline H.; Hayamizu, Terry F.; McCright, Ingeborg J.; Xu, Jingxia; Eppig, Janan T.; Kadin, James<br />

A.; Richardson, Joel E.; Ringwald, Martin (Jackson Lab, USA)<br />

By integrating large amounts of mouse developmental expression in<strong>for</strong>mation, and by making these data readily accessible and easily<br />

searchable, the Gene Expression Database (GXD) supports investigators in their quest to understand the molecular mechanisms of<br />

developmental and disease. GXD contains expression in<strong>for</strong>mation from wild-type and mutant mice and integrates different types of<br />

expression data, including those derived from RNA in situ and immunohistochemistry experiments. Expression data from the<br />

literature is added to the database by the GXD staff. Data is also acquired directly from researchers, including groups doing large-scale<br />

expression studies. GXD currently contains 1.4 million expression results <strong>for</strong> nearly 13,700 genes. As well, GXD has nearly 250,000<br />

images of expression data, allowing users to retrieve the primary data and interpret it themselves. By being an integral part of the<br />

larger Mouse Genome In<strong>for</strong>matics (MGI) resource, GXD combines its expression data with other genetic and disease-oriented data.<br />

Thus, GXD can provide tools that allow users to evaluate expression data in the larger context and search by a wide variety of<br />

parameters and in ways unavailable elsewhere. Recent interface enhancements include the capability to search <strong>for</strong> expression data <strong>for</strong><br />

genes that are associated with specific phenotypes and/or human diseases. Data summaries have become more interactive and include<br />

export features that make it possible to download these data <strong>for</strong> further analyses. GXD is available through the MGI web site at<br />

www.in<strong>for</strong>matics.jax.org/expression.shtml. GXD is supported by NIH grant HD062499.<br />

Program/Abstract # 260<br />

The Sanger Mouse Genetics Project: High Throughput Recessive Lethality and DMDD Screens<br />

Galli, Antonella; Ramirez-Solis, Ramiro; Estabel, Jeanne; White, Jacqui; Tuck, Elizabeth; Jones, Catherine; Green, Angela; Hooks,<br />

Yvette; Souter, Luke; Ryder, Edward; Adams, David (Wellcome Trust Sanger Institute, UK); Mohun, Tim; Wilson, Robert (MRC, UK)<br />

The Sanger Institute Mouse Genetics Project (MGP) is a major contributor in the worldwide ef<strong>for</strong>t to develop mouse models to<br />

understand human genetic diseases. Over the next five years, the MGP aims to generate, cryopreserve and per<strong>for</strong>m primary<br />

phenotypic characterization of over 800 lines of mice . A standardised battery of primary phenotyping tests is per<strong>for</strong>med on all lines<br />

without any prior assumptions about gene function. To date, 37% of the 699 lines studied by the MGP are classified as lethal or subviable<br />

at postnatal day 14 due to non-Mendelian homozygous viability rates. To explore potential defects during embryogenesis, the<br />

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