The role of Drosophila Lamin C in muscle function and gene expression

George Dialynas, Sean Speese, Vivian Budnik, Pamela K. Geyer, Lori L. Wallrath

Research output: Contribution to journalArticle

74 Scopus citations

Abstract

The inner side of the nuclear envelope (NE) is lined with lamins, a meshwork of intermediate filaments that provides structural support for the nucleus and plays roles in many nuclear processes. Lamins, classified as A- or B-types on the basis of biochemical properties, have a conserved globular head, central rod and C-terminal domain that includes an Ig-fold structural motif. In humans, mutations in A-type lamins give rise to diseases that exhibit tissue-specific defects, such as Emery-Dreifuss muscular dystrophy. Drosophila is being used as a model to determine tissue-specific functions of A-type lamins in development, with implications for understanding human disease mechanisms. The GAL4-UAS system was used to express wild-type and mutant forms of Lamin C (the presumed Drosophila A-type lamin), in an otherwise wild-type background. Larval muscle-specific expression of wild type Drosophila Lamin C caused no overt phenotype. By contrast, larval muscle-specific expression of a truncated form of Lamin C lacking the N-terminal head (Lamin C ΔN) caused muscle defects and semi-lethality, with adult 'escapers' possessing malformed legs. The leg defects were due to a lack of larval muscle function and alterations in hormone-regulated gene expression. The consequences of Lamin C association at a gene were tested directly by targeting a Lamin C DNA-binding domain fusion protein upstream of a reporter gene. Association of Lamin C correlated with localization of the reporter gene at the nuclear periphery and gene repression. These data demonstrate connections among the Drosophila A-type lamin, hormone-induced gene expression and muscle function.

Original languageEnglish (US)
Pages (from-to)3067-3077
Number of pages11
JournalDevelopment
Volume137
Issue number18
DOIs
StatePublished - Sep 15 2010

Keywords

  • Drosophila
  • Gene expression
  • Lamins
  • Larval muscle
  • Nuclear envelope

ASJC Scopus subject areas

  • Molecular Biology
  • Developmental Biology

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    Dialynas, G., Speese, S., Budnik, V., Geyer, P. K., & Wallrath, L. L. (2010). The role of Drosophila Lamin C in muscle function and gene expression. Development, 137(18), 3067-3077. https://doi.org/10.1242/dev.048231