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  • 1.
    Munoz-Alarcon, Andres
    et al.
    Södertörn University, School of Life Sciences. Karolinska Institute.
    Pavlovic, Maja
    Södertörn University, School of Life Sciences. Karolinska Institute.
    Wismar, Jasmine
    Max-Planck-Institut für Hirnforschung, Frankfurt, Germany.
    Schmitt, Bertram
    Max-Planck-Institut für Hirnforschung, Frankfurt, Germany.
    Eriksson, Maria
    Karolinska Institute.
    Kylsten, Per
    Södertörn University, School of Life Sciences.
    Dushay, Mitchell S.
    Södertörn University, School of Life Sciences. Uppsala University.
    Characterization of lamin Mutation Phenotypes in Drosophila and Comparison to Human Laminopathies2007In: PLOS ONE, E-ISSN 1932-6203, Vol. 2, no 6, p. e532-Article in journal (Refereed)
    Abstract [en]

    Lamins are intermediate filament proteins that make up the nuclear lamina, a matrix underlying the nuclear membrane in all metazoan cells that is important for nuclear form and function. Vertebrate A-type lamins are expressed in differentiating cells, while B-type lamins are expressed ubiquitously. Drosophila has two lamin genes that are expressed in A-and B-type patterns, and it is assumed that similarly expressed lamins perform similar functions. However, Drosophila and vertebrate lamins are not orthologous, and their expression patterns evolved independently. It is therefore of interest to examine the effects of mutations in lamin genes. Mutations in the mammalian lamin A/C gene cause a range of diseases, collectively called laminopathies, that include muscular dystrophies and premature aging disorders. We compared the sequences of lamin genes from different species, and we have characterized larval and adult phenotypes in Drosophila bearing mutations in the lam gene that is expressed in the B-type pattern. Larvae move less and show subtle muscle defects, and surviving lam adults are flightless and walk like aged wild-type flies, suggesting that lam phenotypes might result from neuromuscular defects, premature aging, or both. The resemblance of Drosophila lam phenotypes to human laminopathies suggests that some lamin functions may be performed by differently expressed genes in flies and mammals. Such still-unknown functions thus would not be dependent on lamin gene expression pattern, suggesting the presence of other lamin functions that are expression dependent. Our results illustrate a complex interplay between lamin gene expression and function through evolution.

  • 2.
    O'Farrell, Fergal
    et al.
    Södertörn University, School of Life Sciences. Karolinska Institutet.
    Munoz-Alarcón, Andrés
    Södertörn University, School of Life Sciences. Karolinska Institutet.
    Georgiev, Alexander
    Stockholms universitet.
    Kylsten, Per
    Södertörn University, School of Life Sciences.
    Functional Analysis of Drosophila lin-41, dappled during adult eye developmentManuscript (preprint) (Other academic)
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  • harvard-anglia-ruskin-university
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  • sodertorns-hogskola-harvard.csl
  • sodertorns-hogskola-oxford.csl
  • Other style
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