BASIC SCIENCE

RESEARCH REPORT FOR JOINT PROJECT

  1. Title of Project:

    The Functions of the ubiquitin ligase, Skp2, and cyclin-dependent kinase 2 (Cdk2) in normal development and cancer

  2. Duration:: April 1, 2006 — March 31, 2007
  3. Project Organization
    1. Japanese Principal Investigator (JPI)
      Dr. Keiichi I. Nakayama (Professor, Dept. of Molecular and Cellular Biology, Medical Institute of Bioregulation, Kyushu University)
    2. U.S. Principal Investigator (USPI)
      Dr. Philipp Kaldis (Principal Investigator, Mouse Cancer Genetics Program, National Cancer Institute
    3. Research Associate (Japanese)
      Syuhei Kotoshiba (Postdoctoral fellow, NCI)
    4. List of Other Project Participants (except for PIs and RA)

      Japanese-side:
      Michiko Shirane (Assistant Professor, Medical Institute of Bioregulation, Kyushu University)
      Masaki Matsumoto (Special Assistant, Medical Institute of Bioregulation, Kyushu University)
      Nishiyama Masaaki (Guest Researcher, Medical Institute of Bioregulation, Kyushu University)
      Sakae Nobutaka (Medical Staff, Kyushu University Hospital)
      Ichiro Onoeyama (Guest Researcher, Medical Institute of Bioregulation, Kyushu University)
      Etsuo Suzaki (JST Dispatch researcher, Medical Institute of Bioregulation, Kyushu University)
      Toru Saiga (Graduate Student, Graduate school of Medicine, Kyushu University)
      Tadashi Nakagawa (Graduate Student, Graduate school of Medicine, Kyushu University)
      Kanae Tanaka (Graduate Student, Graduate school of Medicine, Kyushu University)
      Kouji Oyamada (JST Dispatch engineer, Medical Institute of Bioregulation, Kyushu University)
      Shinji Hatakeyama (Professor, Graduate School of Medicine, Hokkaido University)
      Tadasuke Tsukiyama (Assistant Professor, Graduate School of Medicine, Hokkaido University)
      Taichi Kimura (Contracted Research Associate, Graduate School of Medicine, Hokkaido University)
      Takumi Kamura (Professor, Graduate School of Science, Nagoya University)
      Masatoshi Kitagawa (Professor, School of Medicine, Hamamatsu University)
      Kyoko Kitagawa (Assistant Professor, School of Medicine, Hamamatsu University)
      Ichiro Taniuchi (Team leader, RIKEN Yokohama Institute)
      Kiyotaka Oshikawa (Contracted Research Associate, Medical Institute of Bioregulation, Kyushu University)
      Keiko Nakayama (Professor, Tohoku University School of Medicine)
      Noriko Ishida (Assistant Professor, Tohoku University School of Medicine)
      Kentaro Hara (Researcher, Tohoku University School of Medicine)
      Yoshinori Ishikawa (Graduate Student, Tohoku University School of Medicine)
      Taichi Hara (Researcher, Tokyo Metropolitan Institute of Medical Science, Tokyo Metropolitan Organaization for Medical Research)
      Kensuke Miyamoto (Basic science special researcher, RIKEN Tsukuba Institute)
      Hiroyasu Nagahama (Assistant Professor, Department of Gastroenterology and Hepatology, Kumamoto University of Medicine)
      Ryousuke Tsunematsu (Doctor, Kyushu medical center)
      HidehisaTakahashi (JST Dispatch engineer, Medical Institute of Bioregulation, Kyushu University)
      Hiroshi Fujii (Graduate Student, Graduate school of Medicine, Kyushu University)

      US-side:
      Cyril Berthet (Postdoctoral Fellow, Mouse Cancer Genetics Program, NCI-CCR)
      Eiman Aleem (Postdoctoral Fellow, Mouse Cancer Genetics Program, NCI-CCR)
  4. Activities of Research Associate (RA)
    1. Duration in US: April 1, 2006 ~February 28, 2007
    2. Institution Visited in US:
      Dr. Philipp Kaldis (Mouse Cancer Genetics Program, National Cancer Institute)
    3. Scientific Results/Achievements
      We have set up mouse breedings in order to generate the desired Cdk2/Skp2 DKO mice. We investigated whether they display abnormalities in developmental processes by statistical analysis of litter sizes by determining their genotype. Cdk2/Skp2 DKO mice are viable, even though both Cdk2 and Skp2 are positive regulators of the cell cycle. At present, we got 8 DKO mice with a ratio of 3.29%, compared with wild-type (9.88%), Skp2 KO (3.29%) and Cdk2 KO (6.58%). In addition we prepared mouse embryonic fibroblasts (MEFs) from 5 DKO embryos.
      We analyzed the cell proliferation of these MEFs. Skp2 KO and Cdk2/Skp2 DKO MEFs proliferated at a lower rate than wild-type and Cdk2 KO MEFs. In order to gain insight into the mechanisms underlying the proliferation in these MEFs, we studied the cell cycle kinetics of unsynchronized MEFs. Cells were pulse labeled with BrdU and then subjected to FACS. We observed that the G1 phase was decreased and G2/M phase was increased in Cdk2/Skp2 DKO MEFs. To further characterize the proliferation of these MEFs, we performed Western blotting using these extracts, which showed p27 and cyclin E1 but not cyclin E2 accumulation in Skp2 KO and Cdk2/Skp2 DKO MEFs. In addition, we investigated the composition of the Cdk/cyclin/CKI complexes in lysates from these MEFs. We performed immunoprecipitations with lysates from these MEFs and antibodies against various Cdks and cyclins, then we performed Western blotting. Cdk1 was bound to cyclin E1 in Cdk2 KO, Skp2 KO, and Cdk2/Skp2 DKO but not in wild-type MEFs.
      Large amount of p27was bound to Cdk2 in Skp2 KO MEFs. In addition, we pulled down p27 in the cyclin A2 immunoprecipitates for Skp2 KO and Cdk2/Skp2 DKO MEFs. To measure the kinase activity of Cdks, we performed immunoprecipitations, followed by kinase assays with the substrate histone H1. Cdk2 activity was detected in wild-type MEFs but not in other MEFs. Cyclin A2 activity was detected in each MEFs, but decreased in Cdk2 KO, Skp2 KO, and Cdk2/Skp2 DKO MEFs. Cyclin B1 and Cdk1 activities were decreased in only Skp2 KO MEFs. These results suggest that loss of Cdk2 can rescue reduced Cdk1 and cyclin B1-associated kinase activities in Skp2KO MEFs. We are currently investigating the mechanism behind this.
      Unsynchronized cells do not reveal the timing of the cell cycle progression; therefore we synchronized our MEFs by starvation to study the impact of loss of both Cdk2 and Skp2 on the cell cycle progression. MEFs were plated at high densities, starved for 3 days by culturing in medium supplemented with 0.1% FBS, and then released by addition of 10% FBS. Cells were harvested at several time points after serum stimulation. In addition, MEFs were pulse labeled with BrdU and then subjected to FACS. By 18 hr, a good percentage of wild-type MEFs were in S phase, whereas other MEFs required 6 hr more, and Cdk2/Skp2 DKO MEFs seemed the slowest to enter S phase in these MEFs. We performed Western blotting using these extracts, which showed p27 and cyclin E1 accumulation after 24 hr in both Skp2 KO and Cdk2/Skp2 DKO MEFs and delayed expression of Cdk1 in Cdk2/Skp2 DKO MEFs. To confirm these results, we are doing same experiments with more samples.
      We will monitor the susceptibility to tumors of Cdk2-/-Skp2-/- DKO mice and compare it to Cdk2-/- and Skp2-/- mice. So, we are going to perform colony formation assay using primary MEFs retrovirally transduced with several combinations of oncogenes. We also will use tumor inducers as irradiation and chemical carcinogens.
  5. Number of Exchanges during Project Period
    1. from Japan to US
      Takumi Kamura April.26-May.6.2005 Stowers Institute for Medical Research Cold Spring Harvor Laboratory
      HidehisaTakahashi April.26-May.6.2005 Stowers Institute for Medical Research Cold Spring Harvor Laboratory
      Shuhei Kotoshiba April.26-May.6.2005 Stowers Institute for Medical Research Cold Spring Harvor Laboratory
      Keiichi Nakayama Sep. 5-11 ,2006 LCC / NCI
      Michiko Shirane Sep. 6-11, 2006 NCI
      Masaaki Nishiyama Sep. 6-11, 2006 NCI
      Ichiro Onoyama Sep. 6-11, 2006 NCI
      Toru Saiga Sep. 6-11, 2006 NCI
      Tadashi Nakagawa Sep. 6-11, 2006 NCI
      Masatoshi Kitagawa Sep. 6-11, 2006 NCI
      Ryousuke Tsunematsu Sep. 6-11, 2006 NCI
    2. from US to Japan
      None
  6. Purpose and Content of Project

    Proliferation in normal cells is tightly controlled to ensure one single round of DNA replication and cell division per cell cycle. The activities of cyclin-dependent kinases [Cdks] are thought to be essential for cell cycle progression. For example, in the G1 phase there is minimal Cdk activity, in S phase Cdk activity raises to intermediate levels, and in mitosis [M phase] Cdk activity peaks at high levels. The activity of Cdks is controlled on several levels including binding of subunits [cyclins, Cdk inhibitory molecules {CKI}], phosphorylation, subcellular localization, and transcriptional control. Several Cdk regulatory proteins are short-lived and are degraded by ubiquitin mediated proteolysis. Target proteins are degraded after modification by ubiquitin chains. The recognition of target proteins is governed by ubiquitin ligase enzymes [E3]. Skp2 is one component of an E3 enzyme [SCFSkp2], which targets the CKI p27Kip1 and cyclin E. Both these proteins regulate Cdk2 that in addition binds cyclin A. Cdk2 is the most important target of p27 since overexpression of p27 leads to decreased Cdk2 activity and cell cycle arrest. In cancer, Cdk activity is elevated through mechanisms that are not known yet. Some of the best in vivo models for cancer are mouse models. We are using mouse models to study the functions of Skp2 and Cdk2 during normal development and tumorigenesis. We are proposing to generate double knockout mice that lack both Skp2 and Cdk2. In these mice, there will be no Cdk2 activity and elevated levels of p27 that can affect other targets. With these Skp2-/-Cdk2-/- mice, we are addressing a situation where p27 functions are accentuated [because of the elevated levels] and at the same time there is no Cdk2 that could bind excessive p27. Therefore, the additional p27 molecules will affect targets that could not been uncovered in a wild type background where Cdk2 is the major target of p27. Identification of new unknown targets of p27 is of great interest to understand the functions of p27 in the cell cycle and during tumorigenesis.

    We have generated double knockout mice for Skp2-/-Cdk2-/- and to analyze their phenotype. First, we have analyzed the phenotype of these mice by studying different tissues by histochemistry and biochemistry. In a second stage, we have generated cell models of mouse embryonic fibroblast [MEFs], hepatocytes, thymocytes, and splenocytes. In these cells, we studied the details of cell cycle regulation including the growth characteristics, the kinetics of cell cycle transitions, the activities of Cdks, and the protein complexes formed. In a third stage, we have analyzed the induction of tumors in Skp2-/-Cdk2-/- mice by using a skin and liver tumor model. Tumors will be collected and analyzed for genetic lesions, by (immuno-) histochemistry, and by biochemistry.

    Our project is of great importance not only because it will foster a close collaboration between the Nakayama and Kaldis laboratories but also because of the expected results from this study. Our model is based on a two-strike approach where two pathways [Skp2 and Cdk2] are inactivated simultaneously and the effect on normal development and tumorigenesis is studied. Pharmaceutical companies have developed molecules that inhibit the function of Cdk2 or Skp2. Here we are investigating if it would make sense to combine two different classes of drugs in cancer therapy in an animal model. The outcome of our study has great potential for future cancer therapy as well as basic research of the cell cycle.

  7. Status Report of Project Implementation

    The cyclin-dependent kinase inhibitor p27Kip1 is degraded at the G0-G1 transition of the cell cycle by the ubiquitin-proteasome pathway. Although the nuclear ubiquitin ligase (E3) SCFSkp2 is implicated in p27Kip1 degradation, proteolyis of p27Kip1 at the G0-G1 transition proceeds normally in Skp2–/– cells. Moreover, p27Kip1 is exported from the nucleus to the cytoplasm at G0-G1. These data suggest the existence of a Skp2-independent pathway for the degradation of p27Kip1 at G1 phase. We now describe a previously unidentified E3 complex: KPC (Kip1 ubiquitylation–promoting complex), consisting of KPC1 and KPC2. KPC1 contains a RING-finger domain, and KPC2 contains a ubiquitin-like domain and two ubiquitin-associated domains. KPC interacts with and ubiquitylates p27Kip1 and is localized to the cytoplasm. Overexpression of KPC promoted the degradation of p27Kip1, whereas a dominant-negative mutant of KPC1 delayed p27Kip1 degradation. The nuclear export of p27Kip1 by CRM1 appears to be necessary for KPC-mediated proteolysis. Depletion of KPC1 by RNA interference also inhibited p27Kip1 degradation. KPC thus likely controls degradation of p27Kip1 in G1 phase after export of the latter from the nucleus.

  8. Seminar
    1. Title: "US-Japan Cooperative Research Workshop on Mouse Models for Cell Cycle and Ubiquitin-mediated Degradation"
    2. Period: September 7-8, 2006
    3. Site: National Cancer Institute, Frederick, MD
    4. Participants: Total 19 (Japan side 8 , US side 11 , Other country 0 )
    5. Other Country Participants: None
    6. Agenda, Topics and Scientific Achievements

      September 7, 2006

      8:00am Pickup at the FSK Holiday Inn [I-270] Kaldis lab
      8:20-9:00am Breakfast Boardroom Bldg. 549  
      9:00-9:15am Welcoming address Philipp Kaldis
      9:15-9:45am Fbw7 is a key regulator of cell cycle exit during differentiation Keiichi Nakayama
      9:45-10:00am Characterization of mice that lack Fbw7 in bone marrow, mammary glands or liver Ichiro Onoyama
      10:00-10:30am Genetic analysis of Parc, a cullin family of E3 ubiquitin ligase Yue Xiong
      10:30-10:50 Morning Break  
      10:50-11:05am Fbxo45-PAM, a novel ubiquitin ligase complex, is required for synapse formation Toru Saiga
      11:05-11:20am Rescue of loss of Cdk2 & Cdk4 Padmakumar VC
      11:20-11:35am Fbxw8 is essential for Cul1-Cul7 complex formation and for placental development Ryosuke Tsunematsu
      11:35-11:50am Functions of Rb in the absence of Cdk2 & Cdk4 Weimin Li
      11:50-12:05pm Chromatin remodeling factor CHD8 is a novel oncogene that blocks p53-mediated apoptosis pathway Masaaki Nishiyama
      12:15-1:30pm Lunch  
      1:30-1:45pm DNA Damage in the absence of Cdk2 Satya Ande
      1:45-2:15pm Ubiquitin-mediated regulation of tumor suppressor proteins Masatoshi Kitagawa
      2:15-2:30pm Cdk2 & Cdk4 couple G1/S transition to mitosis Cyril Berthet
      2:30-2:45pm Potential for protein degradation and chromatin cross-talk:Characterization of the nucleolar F-box protein Fbl10 David Frescas
      2:45-3:00pm Interactions of Skp2 and Cdk2 Shuhei Kotoshiba
      3:00-3:15pm Mammalian E4 is required for cardiac development and maintenance of the nervous system Tadashi Nakagawa
      3:15-3:30pm The stability of the tumor suppressor REST/NRSF is controlled by SCF-betaTrcp-mediated degradation Daniele Guaravaccaro
      3:30-4:00pm Afternoon Break  
      4:00-4:15pm Ubiquitin mediated degradation controls Claspin at different phases of the cell cycle Florian Bassermann
      4:15-4:30pm Specific roles for cyclin E1 and E2 Kristy McDowell
      4:30-4:45pm Protrudin interacts with Rab11-GDP and induces neurite formation by directional membrane trafficking Michiko Shirane
      4:45-5:15pm Control of protein translation and cell cycle checkpoints by the F-box protein ßTrcp Michele Pagano
      5:15-5:45pm Cdc2 compensates for loss of Cdk2 Philipp Kaldis
      5:45-6:45pm Round Table Discussions  
      6:45-7:15pm Concluding Remarks Keiichi Nakayama
      7:30pm Dinner followed by Entertainment  

      September 8, 2006

      8:30am Pickup at the FSK Holiday Inn Kaldis lab
      9:00am Breakfast Boardroom 549  
      10:00am Lab tour Bldg. 560  
      10:45am Discussion  
  9. Research Results of Project

    The highly ordered progression of the cell cycle is achieved by a series of elaborate mechanisms that control the periodic expression of many regulatory proteins. One such regulatory protein is the Cdk inhibitor (CKI) p27. In normal cells, the amount of p27 is high during G0 phase but rapidly decreases on the reentry of cells into G1-S. The intracellular concentration of p27 is thought to be regulated predominantly by the ubiquitin-mediated proteolytic pathway. Degradation of p27 is promoted by its phosphorylation on Thr187 by the cyclin E–Cdk2 complex. Skp2, an F-box protein that functions as the receptor component of an SCF ubiquitin ligase complex, binds to p27 only when Thr187 of p27 is phosphorylated; such binding then results in the ubiquitylation and degradation of p27. Skp2 also targets free cyclin E for ubiquitylation. The most obvious cellular phenotype of Skp2–/– mice is the presence of markedly enlarged, polyploid nuclei and multiple centrosomes, suggestive of an impairment in the mechanism that prevents endoreplication, in which the genomic DNA content of a cell is increased without cell division. In addition to p27 and free cyclin E, several other substrates have been proposed for Skp2. We have now generated Skp2–/– p27–/– mice and found that they do not exhibit the overreplication phenotype, suggesting that p27 accumulation is required for its development. Hepatocytes of Skp2–/– mice entered the endoduplication cycle after mitogenic stimulation, whereas this phenotype was not apparent in Skp2–/– p27–/– mice. Cdc2-associated kinase activity was lower in Skp2–/– p27–/– cells than in wild-type cells, and a reduction in Cdc2 activity was sufficient to induce nuclear enlargement and centrosome overduplication. The lack of p27 degradation in G2 phase in Skp2–/– cells may thus result in suppression of Cdc2 activity and consequent inhibition of entry into M phase or reentry into S phase without passage through M phase. These data suggest that Skp2 contributes to regulation of G2-M progression by mediating the degradation of p27.

    In normal cells, the amount of p27 is high during G0 phase of the cell cycle but decreases rapidly on reentry of cells into G1 phase. For example, mitogenic activation of resting lymphocytes or reexposure of serum-deprived embryonic fibroblasts to serum induces rapid degradation of p27 between 3 and 9 h after stimulation. However, Skp2 is not expressed until early S phase (18 to 24 h after stimulation), unequivocally later than the degradation of p27 apparent at G0-G1. Moreover, p27 is exported from the nucleus to the cytoplasm at G0-G1, whereas Skp2 is restricted to the nucleus. These discrepancies between the temporal and spatial patterns of p27 expression and those of Skp2 expression suggested the existence of a Skp2-independent pathway for the degradation of p27. Indeed, the down-regulation of p27 at the G0-G1 transition occurs normally in Skp2–/– cells and is sensitive to proteasome inhibitors, indicating that p27 is degraded at G0-G1 by a proteasome-dependent, but Skp2-independent, mechanism. Biochemical analysis of crude extracts of Skp2–/– cells revealed the presence in the cytoplasmic fraction of a Skp2-independent E3 activity that mediates the ubiquitylation of p27. This ubiquitylation of p27 is not dependent on the phosphorylation of threonine-187, which is a prerequisite for Skp2-mediated ubiquitylation. We have recently purified an E3 enzyme, designated KPC (Kip1 ubiquitylation-promoting complex), that interacts with and ubiquitylates p27 in G1 phase and is localized to the cytoplasm of mammalian cells.

    We also carried out a comprehensive characterization of ubiquitin-conjugated and ubiquitin-associated proteins in mammalian cells. The proteins were purified by immunoaffinity chromatography under denaturing or native conditions. They were then digested with trypsin, and the resulting peptides were analyzed by two-dimensional liquid chromatography and tandem mass spectrometry. A total of 670 distinct proteins was identified; 345 proteins (51%) were classified as Urp-D (ubiquitin-related proteome under the denaturing condition) and comprised ubiquitin-conjugated molecules, whereas 325 proteins (49%) were classified as Urp-N (ubiquitin-related proteome only under the native condition) and included molecules that associated with ubiquitylated proteins. The proportions of proteins in various functional categories differed substantially between Urp-D and Urp-N. Many ribosomal subunits were detected in the Urp-D group of proteins and several of these subunits were directly shown to be ubiquitylated by mass spectrometric analysis, suggesting that ubiquitylation might play an important role in the regulation and/or quality control of ribosomal proteins. Our results demonstrate the potential of proteomics analysis of protein ubiquitylation to provide important insight into the regulation of protein stability and other ubiquitin-related cellular functions.

    We also studied an F-box protein Fbw7, which is structurally similar to Skp2. Cell proliferation is tightly controlled during differentiation. In T cell development, the cell cycle is normally arrested at the CD4+CD8+ stage, but the mechanism underlying such differentiation-specific exit from the cell cycle has been unclear. Fbw7, an F-box protein subunit of an SCF-type ubiquitin ligase complex, induces the degradation of positive regulators of the cell cycle, such as c-Myc, c-Jun, cyclin E and Notch. FBW7 is often mutated in a subset of human cancers. We have now achieved conditional inactivation of the Fbw7 gene in the T cell lineage of mice and found that the cell cycle is not arrested at the CD4+CD8+ stage in the homozygous mutant animals. The mutant mice manifested thymic hyperplasia likely through c-Myc accumulation and eventually developed thymic lymphoma. In contrast, mature T cells of the mutant mice failed to proliferate in response to mitogenic stimulation and underwent apoptosis in association with accumulation of c-Myc and p53. These abnormalities were corrected by deletion of p53. Our results suggest that Fbw7 regulates the cell cycle in a differentiation-dependent manner, its loss resulting in c-Myc accumulation that leads to hyperproliferation in immature T cells but to p53-dependent cell cycle arrest and apoptosis in mature T cells.

  10. Papers and Publications (Project-related papers that have or will be published)

    Rodier, G., Makris, C., Coulombe, P., Scime, A., Nakayama, K., Nakayama, K.I., Meloche, S.: p107 inhibits G1 to S phase progression by down-regulating expression of the F-box protein Skp2. J. Cell Biol., 168: 55-66 (2005).

    Kotake, Y., Nakayama, K., Ishida, N., Nakayama, K.I.: Role of serine 10 phosphorylation in p27 stabilization revealed by analysis of p27 knock-in mice harboring a serine 10 mutation. J. Biol. Chem., 280: 1095-1102 (2005).

    Harada, K., Takeuchi, H., Oike, M., Matsuda, M., Kanematsu, T., Yagisawa, H., Nakayama, K.I., Maeda, K., Erneux, C., Hirata, M.: Role of PRIP-1, a novel Ins(1,4,5)P(3) binding protein, in Ins(1,4,5)P(3)-mediated Ca(2+) signaling. J. Cell. Physiol., 202: 422-433 (2005).

    Uchida, T., Nakamura, T., Hashimoto, N., Matsuda, T., Kotani, K., Sakaue, H., Kido, Y., Hayashi, Y., Nakayama, K.I., White, M. F., Kasuga, M.: Deletion of Cdkn1b ameliorates hyperglycemia by maintaining compensatory hyperinsulinemia in diabetic mice. Nature Med., 11: 175-182 (2005).

    Takahashi, H., Hatakeyama, S., Saitoh, H., Nakayama, K.I.: Noncovalent SUMO-1 binding activity of thymine DNA glycosylase (TDG) is required for its SUMO-1 modification and colocalization with the promyelocytic leukemia protein. J. Biol. Chem., 280: 5611-5621 (2005).

    He, C. H., Waxman, A. B., Lee, C. G., Link, H., Rabach, M. E., Ma, B., Chen, Q., Zhu, Z., Zhong, M., Nakayama, K., Nakayama, K.I., Homer, R., Elias, J. A.: Bcl-2-related protein A1 is an endogenous and cytokine-stimulated mediator of cytoprotection in hyperoxic acute lung injury. J. Clin. Invest., 115: 1039-1048 (2005).

    Jackson, D., Zheng, Y., Lyo, D., Shen, Y., Nakayama, K., Nakayama, K.I., Humphries, M. J., Reyland, M. E., Foster, D. A.: Suppression of cell migration by protein kinase Cdelta. Oncogene, 24: 3067-3072 (2005).

    Kotoshiba, S., Kamura, T., Hara, T., Ishida, N., Nakayama, K.I.: Molecular dissection of the interaction between p27 and Kip1 ubiquitylation-promoting complex, the ubiquitin ligase that regulates proteolysis of p27 in G1 phase. J. Biol. Chem., 280: 17694-17700 (2005).

    Kase, S., Yoshida, K., Ikeda, H., Harada, T., Harada, C., Imaki, J., Ohgami, K., Shiratori, K., Nakayama, K.I., Nakayama, K., Ohno, S.: Disappearance of p27(KIP1) and increase in proliferation of the lens cells after extraction of most of the fiber cells of the lens. Curr. Eye Res., 30: 437-442 (2005).

    Jiang, H., Chang, F. C., Ross, A. E., Lee, J., Nakayama, K.I., Nakayama, K., Desiderio, S.: Ubiquitylation of RAG-2 by Skp2-SCF links destruction of the V(D)J recombinase to the cell cycle. Mol. Cell, 18: 699-709 (2005).

    Pushkarsky, T., Yurchenko, V., Vanpouille, C., Brichacek, B., Vaisman, I., Hatakeyama, S., Nakayama, K.I., Sherry, B., Bukrinsky, M. I.: Cell surface expression of CD147/EMMPRIN is regulated by cyclophilin 60. J. Biol. Chem., 280: 27866-27871 (2005).

    Wang, H. Q., Nakaya, Y., Du, Z., Yamane, T., Shirane, M., Kudo, T., Takeda, M., Takebayashi, K., Noda, Y., Nakayama, K.I., Nishimura, M.: Interaction of presenilins with FKBP38 promotes apoptosis by reducing mitochondrial Bcl-2. Hum. Mol. Genet., 14: 1889-1902 (2005).

    Kase, S., Yoshida, K., Nakayama, K.I., Nakayama, K., Ikeda, H., Harada, T., Harada, C., Ohgami, K., Shiratori, K., Ohno, S.: Phosphorylation of p27(KIP1) in the developing retina and retinoblastoma. Int. J. Mol. Med., 16: 257-262 (2005).

    Chen, Z., Foster, M. W., Zhang, J., Mao, L., Rockman, H. A., Kawamoto, T., Kitagawa, K., Nakayama, K.I., Hess, D. T., Stamler, J. S.: An essential role for mitochondrial aldehyde dehydrogenase in nitroglycerin bioactivation. Proc. Natl. Acad. Sci. U. S. A., 102: 12159-12164 (2005).

    Hatakeyama, S., Watanabe, M., Fujii, Y., Nakayama, K.I.: Targeted destruction of c-Myc by an engineered ubiquitin ligase suppresses cell transformation and tumor formation. Cancer Res., 65: 7874-7879 (2005).

    Hino, S., Tanji, C., Nakayama, K.I., Kikuchi, A.: Phosphorylation of beta-catenin by cyclic AMP-dependent protein kinase stabilizes beta-catenin through inhibition of its ubiquitination. Mol. Cell. Biol., 25: 9063-9072 (2005).

    Hara, T., Kamura, T., Kotoshiba, S., Takahashi, H., Fujiwara, K., Onoyama, I., Shirakawa, M., Mizushima, N., Nakayama, K.I.: Role of the UBL-UBA protein KPC2 in degradation of p27 at G1 phase of the cell cycle. Mol. Cell. Biol., 25: 9292-9303 (2005).

    Matsumoto, M., Hatakeyama, S., Oyamada, K., Oda, Y., Nishimura, T., Nakayama, K.I.: Large-scale analysis of the human ubiquitin-related proteome. Proteomics, 5: 4145-4151 (2005).

    Kaneko-Oshikawa, C., Nakagawa, T., Yamada, M., Yoshikawa, H., Matsumoto, M., Yada, M., Hatakeyama, S., Nakayama, K., Nakayama, K.I.: Mammalian E4 is required for cardiac development and maintenance of the nervous system. Mol. Cell. Biol., 25: 10953-10964 (2005).

    Yogosawa, S., Hatakeyama, S., Nakayama, K.I., Miyoshi, H., Kohsaka, S., Akazawa, C.: Ubiquitylation and degradation of serum-inducible kinase by hVPS18, a RING-H2 type ubiquitin ligase. J. Biol. Chem., 280: 41619-41627 (2005).

    Nishitani, H., Sugimoto, N., Roukos, V., Nakanishi, Y., Saijo, M., Obuse, C., Tsurimoto, T., Nakayama, K.I., Nakayama, K., Fujita, M., Lygerou, Z., Nishimoto, T.: Two E3 ubiquitin ligases, SCF-Skp2 and DDB1-Cul4, target human Cdt1 for proteolysis. EMBO J., 25: 1126-1136 (2006).

    Kase, S., Yoshida, K., Ohgami, K., Shiratori, K., Suzuki, Y., Nakayama, K.I., Ohno, S.: Expression of cdc2 and p27(KIP1) phosphorylation in mitotic cells of the human retinoblastoma. Int. J. Mol. Med., 17: 465-468 (2006).

    Iwanaga, R., Komori, H., Ishida, S., Okamura, N., Nakayama, K., Nakayama, K.I., Ohtani, K.: Identification of novel E2F1 target genes regulated in cell cycle-dependent and independent manners. Oncogene, 25: 1786-1798 (2006).

    Kase, S., Yoshida, K., Harada, T., Harada, C., Namekata, K., Suzuki, Y., Ohgami, K., Shiratori, K., Nakayama, K.I., Ohno, S.: Phosphorylation of extracellular signal-regulated kinase and p27(KIP1) after retinal detachment. Graefes Arch. Clin. Exp. Ophthalmol., 244: 352-358 (2006).

    Sugihara, E., Kanai, M., Saito, S., Nitta, T., Toyoshima, H., Nakayama, K., Nakayama, K.I., Fukasawa, K., Schwab, M., Saya, H., Miwa, M.: Suppression of centrosome amplification after DNA damage depends on p27 accumulation. Cancer Res., 66: 4020-4029 (2006).

    Ryer, E. J., Hom, R. P., Sakakibara, K., Nakayama, K.I., Nakayama, K., Faries, P. L., Liu, B., Kent, K. C.: PKCdelta is necessary for Smad3 expression and transforming growth factor beta-induced fibronectin synthesis in vascular smooth muscle cells. Arterioscler. Thromb. Vasc. Biol., 26: 780-786 (2006).

    Humphries, M. J., Limesand, K. H., Schneider, J. C., Nakayama, K.I., Anderson, S. M., Reyland, M. E.: Suppression of apoptosis in the protein kinase Cdelta null mouse in vivo. J. Biol. Chem., 281: 9728-9737 (2006).

    Kase, S., Yoshida, K., Ohgami, K., Shiratori, K., Ohno, S., Nakayama, K.I.: Phosphorylation of p27(KIP1) in the mitotic cells of the corneal epithelium. Curr. Eye Res., 31: 307-312 (2006).

    Parcellier, A., Brunet, M., Schmitt, E., Col, E., Didelot, C., Hammann, A., Nakayama, K., Nakayama, K.I., Khochbin, S., Solary, E., Garrido, C.: HSP27 favors ubiquitination and proteasomal degradation of p27Kip1 and helps S-phase re-entry in stressed cells. FASEB J., (2006).

    Fotovati, A., Nakayama, K., Nakayama, K.I.: Impaired germ cell development due to compromised cell cycle progression in Skp2-deficient mice. Cell Div., 1: 4 (2006).

    Yoshida, K., Yamaguchi, T., Shinagawa, H., Taira, N., Nakayama, K.I., Miki, Y.: Protein kinase C {delta} activates topoisomerase II{alpha} to induce apoptotic cell death in response to DNA damage. Mol. Cell. Biol., 26: 3414-3431 (2006).

    Niki, S., Oshikawa, K., Mouri, Y., Hirota, F., Matsushima, A., Yano, M., Han, H., Bando, Y., Izumi, K., Matsumoto, M., Nakayama, K.I., Kuroda, N., Matsumoto, M.: Alteration of intra-pancreatic target-organ specificity by abrogation of Aire in NOD mice. J. Clin. Invest., 116: 1292-1301 (2006).

    Shukla, A., Barrett, T. F., Nakayama, K.I., Nakayama, K., Mossman, B. T., Lounsbury, K. M.: Transcriptional up-regulation of MMP12 and MMP13 by asbestos occurs via a PKCdelta-dependent pathway in murine lung. FASEB J., 20: 997-999 (2006).

    Nojima, T., Hayashi, K., Goitsuka, R., Nakayama, K., Nakayama, K.I., Kitamura, D.: Double knockout mice show BASH and PKCdelta have different epistatic relationships in B cell maturation and CD40-mediated activation. Immunol. Lett., 105: 48-54 (2006).

    Shimazu, T., Komatsu, Y., Nakayama, K.I., Fukazawa, H., Horinouchi, S., Yoshida, M.: Regulation of SV40 large T-antigen stability by reversible acetylation. Oncogene, (2006).

    Tsukuba, T., Yamamoto, S., Yanagawa, M., Okamoto, K., Okamoto, Y., Nakayama, K.I., Kadowaki, T., Yamamoto, K.: Cathepsin E-deficient mice show increased susceptibility to bacterial infection associated with the decreased expression of multiple cell surface Toll-like receptors. J. Biochem., 140: 57-66 (2006).

    Tsunematsu, R., Nishiyama, M., Kotoshiba, S., Saiga, T., Kamura, T., Nakayama, K.I.: Fbxw8 is essential for Cul1-Cul7 complex formation and for placental development. Mol. Cell. Biol., 26: 6157-6169 (2006).

    Fujii, Y., Yada, M., Nishiyama, M., Kamura, T., Takahashi, H., Tsunematsu, R., Susaki, E., Nakagawa, T., Matsumoto, A., Nakayama, K.I.: Fbxw7 contributes to tumor suppression by targeting multiple proteins for ubiquitin-dependent degradation. Cancer Sci., 97: 729-736 (2006).

    Kanematsu, T., Yasunaga, A., Mizoguchi, Y., Kuratani, A., Kittler, J. T., Jovanovic, J. N., Takenaka, K., Nakayama, K.I., Fukami, K., Takenawa, T., Moss, S. J., Nabekura, J., Hirata, M.: Modulation of GABA(A) receptor phosphorylation and membrane trafficking by phospholipase C-related inactive protein/protein phosphatase 1 and 2A signaling complex underlying brain-derived neurotrophic factor-dependent regulation of GABAergic inhibition. J. Biol. Chem., 281: 22180-22189 (2006).

    Hiramatsu, Y., Kitagawa, K., Suzuki, T., Uchida, C., Hattori, T., Kikuchi, H., Oda, T., Hatakeyama, S., Nakayama, K.I., Yamamoto, T., Konno, H., Kitagawa, M.: Degradation of Tob1 mediated by SCFSkp2-dependent ubiquitination. Cancer Res., 66: 8477-8483 (2006).

    Matsumoto, A., Onoyama, I., Nakayama, K.I.: Expression of mouse Fbxw7 isoforms is regulated in a cell cycle- or p53-dependent manner. Biochem. Biophys. Res. Commun., 350: 114-119 (2006).

    Shirane, M., Nakayama, K.I.: Protrudin induces neurite formation by directional membrane trafficking. Science, 314: 818-821 (2006).

    Hara, K., Nakayama, K.I., Nakayama, K.: Geminin is essential for the development of preimplantation mouse embryos. Genes Cells, 11: 1281-1293 (2006).

    Takahashi, A., Ohtani, N., Yamakoshi, K., Iida, S., Tahara, H., Nakayama, K., Nakayama, K.I., Ide, T., Saya, H., Hara, E.: Mitogenic signalling and the p16INK4a-Rb pathway cooperate to enforce irreversible cellular senescence. Nature Cell Biol., 8: 1291-1297 (2006).

    Gao, Y., Kitagawa, K., Hiramatsu, Y., Kikuchi, H., Isobe, T., Shimada, M., Uchida, C., Hattori, T., Oda, T., Nakayama, K., Nakayama, K.I., Tanaka, T., Konno, H., Kitagawa, M.: Up-regulation of GPR48 induced by down-regulation of p27Kip1 enhances carcinoma cell invasiveness and metastasis. Cancer Res., 66: 11623-11631 (2006).

    Pula, G., Schuh, K., Nakayama, K., Nakayama, K.I., Walter, U., Poole, A. W.: PKCdelta regulates collagen-induced platelet aggregation through inhibition of VASP-mediated filopodia formation. Blood, 108: 4035-4044 (2006).

    Shukla, A., Lounsbury, K. M., Barrett, T. F., Gell, J., Rincon, M., Butnor, K. J., Taatjes, D. J., Davis, G. S., Vacek, P., Nakayama, K.I., Nakayama, K., Steele, C., Mossman, B. T.: Asbestos-induced peribronchiolar cell proliferation and cytokine production are attenuated in lungs of protein kinase C-{delta} knockout mice. Am. J. Pathol., 170: 140-151 (2007).

    Tu, X., Joeng, K. S., Nakayama, K.I., Nakayama, K., Rajagopal, J., Carroll, T. J., McMahon, A. P., Long, F.: Noncanonical Wnt signaling through G protein-linked PKCdelta activation promotes bone formation. Dev. Cell, 12: 113-127 (2007).

    Yanagawa, M., Tsukuba, T., Nishioku, T., Okamoto, Y., Okamoto, K., Takii, R., Terada, Y., Nakayama, K.I., Kadowaki, T., Yamamoto, K.: Cathepsin E deficiency induces a novel form of lysosomal storage disorder showing the accumulation of lysosomal membrane sialoglycoproteins and the elevation of lysosomal pH in macrophages. J. Biol. Chem., 282: 1851-1862 (2007).

    Itoh, Y., Masuyama, N., Nakayama, K., Nakayama, K.I., Gotoh, Y.: The cyclin-dependent kinase inhibitors p57 and p27 regulate neuronal migration in the developing mouse neocortex. J. Biol. Chem., 282: 390-396 (2007).

    Sakai, T., Sakaue, H., Nakamura, T., Okada, M., Matsuki, Y., Watanabe, E., Hiramatsu, R., Nakayama, K., Nakayama, K.I., Kasuga, M.: Skp2 controls adipocyte proliferation during the development of obesity. J. Biol. Chem., 282: 2038-2046 (2007).

    Uchida, T., Iwashita, N., Ohara-Imaizumi, M., Ogihara, T., Nagai, S., Choi, J. B., Tamura, Y., Tada, N., Kawamori, R., Nakayama, K.I., Nagamatsu, S., Watada, H.: Protein kinase Cdelta plays a non-redundant role in insulin secretion in pancreatic beta cells. J. Biol. Chem., 282: 2707-2716 (2007).

    Mizokami, A., Kanematsu, T., Ishibashi, H., Yamaguchi, T., Tanida, I., Takenaka, K., Nakayama, K.I., Fukami, K., Takenawa, T., Kominami, E., Moss, S. J., Yamamoto, T., Nabekura, J., Hirata, M.: Phospholipase C-related inactive protein is involved in trafficking of gamma2 subunit-containing GABA(A) receptors to the cell surface. J. Neurosci., 27: 1692-1701 (2007).

    Matsuda, T., Matsumoto, A., Uchida, M., Kanaly, R., Misaki, K., Shibutani, S., Kawamoto, T., Kitagawa, K., Nakayama, K.I., Tomokuni, K., Ichiba, M.: Increased formation of hepatic N2-ethylidene-2'-deoxyguanosine DNA adducts in aldehyde dehydrogenase 2 knockout mice treated with ethanol. Carcinogenesis, (2007).

    Liu, Z., Liu, X., Nakayama, K.I., Nakayama, K., Ye, K.: Protein kinase C-delta phosphorylates Ebp1 and prevents its proteolytic degradation, enhancing cell survival. J. Neurochem., 100: 1278-1288 (2007).

    Moller, C., Karlberg, M., Abrink, M., Nakayama, K.I., Motoyama, N., Nilsson, G.: Bcl-2 and Bcl-XL are indispensable for the late phase of mast cell development from mouse embryonic stem cells. Exp. Hematol., 35: 385-393 (2007).

    Nakagawa, T., Shirane, M., Iemura, S., Natsume, T., Nakayama, K.I.: Anchoring of the 26S proteasome to the organellar membrane by FKBP38. Genes Cells, in press.

    Miyamoto, K., Araki, K.Y., Naka, K., Arai, F., Takubo, K., Yamazaki, S., Matsuoka, S., Miyamoto, T., Ito, K., Ohmura, M., Chen, C., Hosokawa, K., Nakauchi, H., Nakayama, K., Nakayama, K.I., Harada, M., Motoyama, N., Suda, T., Hirao, A. Foxo3a is essential for maintenance of the hematopoietic stem cell pool. Cell Stem Cell, in press.

    Susaki, E., Nakayama, K., Nakayama, K.I.: Cyclin D2 translocates p27 out of the nucleus and promotes its degradation at the G0-G1 transition. Mol. Cell. Biol., in press.

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