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  • Živalski modeli v genskem zdravljenju cistične fibroze = Animal models in gene therapy of cystic fibrosis
    Ravnik-Glavač, Metka ; Glavač, Damjan
    Background. Cystic fibrosis (CF) is one of the most common monogenic disease in white population. The ultimate incurability of this lethal disease calls for novel therapeutic strategies, among them ... gene therapy is one of the most promising. As the respiratory consequences of the disease are largely responsible for the mortality of the CF, the lungs are the obvious target sitefor gene therapy. Several problems related to gene transfer have arised concerning the efficiency of transfer, immune response to viral vectors, and the choose of the most relevant lung cells to target. To achieve a progress inCF gene therapy better understanding of fundamental principles of the disease and of lung physiology are needed for what appropriate animal models are crucial. Conclusions. Animal models of cystic fibrosis are represented. The mouse model of CF generated by homologeous recombination is at the moment the only existing animal model with mutated Cftr gene. However, CF mouse didn't develop lung disease, the most characteristic feature of CF in humans. The ovine model of CF would possibly be more appropriate model for studing gene transfer techniques and for evaluating of efficiency and safety, because of the greather similarity in anathomy and function of ovine and human lungs. Ovine model of CF could be produced from naturally cftr mutated sheep or with homologeous recombination of embrional stem cells and cloning. Although monkey's stem cell lines have been developed it will probably take some time before lage animal models of human genetic diseases will be generated. For this reason we have developed an alternative strategy to produce natural animal model of recessive disease based on genetic analysis of genomic DNA. Weintroduce our approach for searching of natural heterozygotes for cftr mutation in monkeys.(Abstract truncated at 2000 characters.)
    Vir: Zdravniški vestnik : glasilo Slovenskega zdravniškega društva = Slovenian medical journal = journal of Slovenian Medical Association. - ISSN 1318-0347 (Letn. 68, št. 11, nov. 1999, str. 673-679)
    Vrsta gradiva - članek, sestavni del
    Leto - 1999
    Jezik - slovenski
    COBISS.SI-ID - 10993369

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