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Investigation of disturbances in copper metabolism on iron homeostasis in animal models of Menkes and Wilson diseaseearly in the early postnatal life.

2012/05/B/NZ4/02423

Keywords

Descriptors

  • NZ4_11: Metabolizm

Panel

NZ4 – Biology of tissues, organs and organisms: morphology and functions of animal's and human's systems, organs and organisms, experimental medicine, basics of neurology

Host institution

Uniwersytet Jagielloński, Wydział Biologii i Nauk o Ziemi

Voivodeship: małopolskie

Other projects carried out by the institution »

Principal investigator (from the host institution)

dr hab. Małgorzata Danuta Lenartowicz

Number of co-investigators in the project: 4

Call

OPUS 3 – announced on 2012-03-15

Amount awarded
539 700 PLN
Project start date
2013-02-13
Project end date
2017-02-12
Project duration
48 months (the same as in the proposal)
Project status
Project settled

Equipment purchased [PL]

  1. NanoDrop 2000 Spektrofotometr. Cost: 60 000 PLN

Information in the final report

Publication in academic press/journals (9)

  1. Metabolizm miedzi oraz charakterystyka dziedzicznych zespołów chorobowych na tle niedoboru miedzi, spowodowanych zaburzeniami aktywnosci białka ATP7A.

    Authors:
    Krzeptowski W, Pierzchała O, Lenartowicz M
    Academic press:
    Kosmos (year: 2014, volume: 63, pages: 395-413), Publisher: Polskie Towarzystwo Przyrodników im. Kopernika
    Status:
    Published
    DOI:
    link to the publication

  2. Haemolysis and perturbations in the systemic iron metabolism of suckling, copper-deficient mosaic mutant mice - an animal model of Menkes disease

    Authors:
    Lenartowicz M, Starzyński RR, Krzeptowski W, Grzmil P, Bednarz A, Ogórek M, Pierzchała O, Staroń R, Gajowiak A, Lipiński P.
    Academic press:
    Plos One (year: 2014, volume: Sep 23;9(9), pages: e107641), Publisher: Public Library of Science
    Status:
    Published
    DOI:
    10.1371/journal.pone.0107641 – link to the publication

  3. Mottled Mice and Non-Mammalian Models of Menkes Disease.

    Authors:
    Lenartowicz M, Krzeptowski W, Lipiński P, Grzmil P, Starzyński R, Pierzchała O, Møller LB.
    Academic press:
    Frontiers in Molecular Neuroscience (year: 2015, volume: 8, pages: eCollection 2015, 8:72), Publisher: Frontiers Media SA Production Office Science Park PSE-D1015 Lausanne, Vaud, Szwajcaria
    Status:
    Published
    DOI:
    10.3389/fnmol.2015.00072 – link to the publication

  4. Copper therapy reduces intravascular hemolysis and derepresses ferroportin in mice with mosaic mutation (Atp7amo-ms): an implication for copper-mediated regulation of the Slc40a1 gene expression.

    Authors:
    Lenartowicz M, Starzyński RR, Jończy A, Staroń R, Antoniuk J, Krzeptowski W, Grzmil P, Bednarz A, Pierzchała O, Ogórek M, Rajfur Z, Baster Z, Lipiński P.
    Academic press:
    Biochimica et Biophysica Acta - Molecular Basis of Disease (year: 2017, volume: pii:S0925-4439, pages: pii:S0925-4439(17)30069-8), Publisher: ELSEVIER
    Status:
    Published
    DOI:
    10.1016/j.bbadis.2017.02.020 – link to the publication

  5. Budowa i funkcja białek z rodziny CTR- błonowych transporterów jednowartosciowych jonów metali.

    Authors:
    Pierzchała O, Ogórek M, Bednarz A, Lenartowicz M
    Academic press:
    Postępy Biologii Komórki (year: 2015, volume: 42, pages: 351-373), Publisher: Fundacja Biologii Komórki i Biologii Molekularnej, ul. Marymoncka 99, 01-813 Warszawa
    Status:
    Published
    DOI:
    link to the publication

  6. Functional iron deficiency in toxic milk mutant mice (tx-J) despite high hepatic ferroportin: a critical role of decreased GPI-ceruloplasmin expression in liver macrophages

    Authors:
    Aneta Jończy, Paweł Lipiński, Mateusz Ogórek, Rafał Starzyński, Daria Krzysztofik, Aleksandra Bednarz , Wojciech Krzeptowski, Mateusz Szudzik, Olga Haberkiewicz, Agnieszka Miłoń, Paweł Grzmil, Małgorzata Lenartowicz
    Academic press:
    Metallomics (year: 2019, volume: 11 (6), pages: 1079-1092.), Publisher: Journal of Royal Society of Chemistry (GB)
    Status:
    Published
    DOI:
    10.1039/c9mt00035f – link to the publication

  7. Atp7a and Atp7b regulate copper homeostasis in developing male germ cells in mice.

    Authors:
    Mateusz Ogórek, Małgorzata Lenartowicz, Rafał Starzyński, Aneta Jończy, Robert Staroń, Andrzej Doniec, Wojciech Krzeptowski, Aleksandra Bednarz, Olga Pierzchała, Paweł Lipiński, Zenon Rajfur, Zbigniew Baster, Paweł Grzmil
    Academic press:
    Metallomics (year: 2017, volume: 9, pages: 1288-1303), Publisher: Journal of Royal Society of Chemistry (GB)
    Status:
    Published
    DOI:
    10.1039/c7mt00134g. – link to the publication

  8. Molecular machinery providing copper bioavailability for spermatozoa along the epididymial tubule in mouse

    Authors:
    Ogórek M, Herman S, Pierzchała O, Bednarz A, Rajfur Z, Baster Z, Grzmil P, Starzyński RR, Szudzik M, Jończy A, Lipiński P, Lenartowicz M
    Academic press:
    Biology of Reproduction (year: 2019, volume: 100 (6), pages: 1505-1520), Publisher: Oxford Academic
    Status:
    Published
    DOI:
    10.1093/biolre/ioz028 – link to the publication

  9. Rola miedzi w procesie spermatogenezy

    Authors:
    Mateusz Ogórek, Łukasz Gąsior, Olga Pierzchała, Regina Daszkiewicz, Małgorzata Lenartowicz
    Academic press:
    Postępy higieny i medycyny Doswiadczalnej (year: 2017, volume: 71, pages: 663-683), Publisher: Czasopismo Instytutu Immunologii i Terapii Doświadczalnej Polskiej Akademii Nauk im. Ludwika Hirszfelda
    Status:
    Published
    DOI:
    10.5604/01.3001.0010.3846 – link to the publication

Articles in post-conference publications (8)

  1. Key role of copper in spermatogenesis

    Authors:
    Ogórek M, Grzmil P, Pierzchała O, Bednarz A, Staroń R, Starzyński R, Lenartowicz M
    Conference:
    2nd Meeting of the Polish Iron Club (year: 2015, pages: 21), Publisher: Postępy Biologii Komórki, Fundacja Biologii Komórki i Biologii Molekularnej, ul. Marymoncka 99, 01-813 Warszawa
    Date:
    conference 12-13 maj
    Status:
    Published

  2. Decrease in SOD1 activity leads to haemolysis and iron metabolism disturbances in mosaic mutant mice (Atp7a mo-ms), an animal model of Menkes disease.

    Authors:
    Małgorzata Lenartowicz, Starzyński Rafał R., Krzeptowski Wojciech, Grzmil Paweł, Bednarz Aleksandra, Ogorek Mateusz, Pierzchała Olga, Staroń Robert, Gajowiak Anna, Lipiński Paweł.
    Conference:
    The Europen Iron Club Meeting 2014, Verona, Italy (year: 2014, pages: 70), Publisher: The Europen Iron Club For Professionals in Biomedical Inorganic Iron
    Date:
    conference 11-14 wrzesien
    Status:
    Published
    DOI:
    link to the publication

  3. Identification of reticuloendothelial cells in the kidneys of young mice and their plausible function in iron metabolism.

    Authors:
    Bednarz A, Lipiński P, Lenartowicz M
    Conference:
    2nd Meeting of the Polish Iron Club (year: 2015, pages: 13), Publisher: Postępy Biologii Komórki, Fundacja Biologii Komórki i Biologii Molekularnej, ul. Marymoncka 99, 01-813 Warszawa
    Date:
    conference 12-13 maj
    Status:
    Published

  4. Regulation of ferroportin expression by copper: studies on mosaic mutant mice – an animal models of Menkes disease.

    Authors:
    Jończy Aneta , Małgorzata Lenartowicz, Rafał R. Starzyński, Robert Staroń, Justyna Antoniuk, Wojciech Krzeptowski, Paweł Grzmil, Aleksandra Bednarz, Olga Pierzchała, Mateusz Ogórek, Paweł Lipiński
    Conference:
    10th International Copper Research Meeting (year: 2016, pages: 92), Publisher: Organizers and Committees of 10th International Copper Research Meeting
    Date:
    conference 25-30 wrzesień
    Status:
    Published

  5. Characterization of iron metabolism related genes in the Jackson Laboratory toxic milk mouse model of Wilson disease

    Authors:
    Aneta JończyJ, Paweł Lipiński, Mateusz Oqórek, Rafał R. Starzyński, Mateusz Szudzik, Olga Pierzchała. Aleksandra Bednarz, Patrycja Kurowska,urowska-, Agnieszka Miłoń, Małgorzata Lenartowicz
    Conference:
    3rd Meeting of the Polish Iron Club Gdańsk, Poland, 15-16 September, 2017 (year: 2017, pages: 6), Publisher: Acta Biochimica Polonica The Journal of the Polish Biochemical Society and of the Committee of Biochemistry and Biophysics Polish Academy of Sciences
    Date:
    conference 15-16 September, 2017
    Status:
    Published

  6. Impact of copper deficiency on the expression of the gene Ctr1 and cellular localisation of CTR1 protein in mice with mutation in the Atp7a gene

    Authors:
    Pierzchała O, Bednarz A, Krzeptowski W, Lenartowicz M
    Conference:
    2nd Meeting of the Polish Iron Club (year: 2015, pages: 27), Publisher: Postępy Biologii Komórki, Fundacja Biologii Komórki i Biologii Molekularnej, ul. Marymoncka 99, 01-813 Warszawa
    Date:
    conference 12-13 maj
    Status:
    Published

  7. Wykorzystanie metod histochemicznych dla wykazania akumulacji żelaza i miedzi w komórkach wątroby myszy mutantów Atp7b (tx-j)- mysiego modelu choroby Wilsona

    Authors:
    Krzysztofik D, Bednarz A, Ogórek M, Jończy A, Lenartowicz M
    Conference:
    XXXII Ogólnopolski Seminarium Mechanizmy służące utrzymaniu życia i regulacji fizjologicznych (year: 2018, pages: 21-22), Publisher: Małopolska Wyższa Szkoła im. J. Dietla w Krakowie
    Date:
    conference czerwiec 2018
    Status:
    Published

  8. Pathological and adaptative changes in the kidney of the mottled mutants as an effect of copper supplementation therapy.

    Authors:
    1.Olga Pierzchała, Aneta Jończy, Rafał R. Starzyński, Robert Staroń, Mateusz Ogórek, Wojciech Krzeptowski, Paweł Grzmil, Aleksandra Bednarz, Paweł Lipiński, Małgorzata Lenartowicz.
    Conference:
    10th International Copper Research Meeting (year: 2016, pages: 106), Publisher: Organizers and Committees of 10th International Copper Research Meeting
    Date:
    conference 25-30 września
    Status:
    Published

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Graphic design and implementation: Jakub Piotrowski