Drosophila melanogaster as a Model to Study Fragile X-Associated Disorders

Jelena Trajković, Vedrana Makevic, Milica Pesic, Sofija Pavković-Lučić, Sara Milojevic, Smiljana Cvjetkovic, Randi Hagerman, Dejan B. Budimirovic, Dragana Protic

Research output: Contribution to journalReview articlepeer-review


Fragile X syndrome (FXS) is a global neurodevelopmental disorder caused by the expansion of CGG trinucleotide repeats (≥200) in the Fragile X Messenger Ribonucleoprotein 1 (FMR1) gene. FXS is the hallmark of Fragile X-associated disorders (FXD) and the most common monogenic cause of inherited intellectual disability and autism spectrum disorder. There are several animal models used to study FXS. In the FXS model of Drosophila, the only ortholog of FMR1, dfmr1, is mutated so that its protein is missing. This model has several relevant phenotypes, including defects in the circadian output pathway, sleep problems, memory deficits in the conditioned courtship and olfactory conditioning paradigms, deficits in social interaction, and deficits in neuronal development. In addition to FXS, a model of another FXD, Fragile X-associated tremor/ataxia syndrome (FXTAS), has also been established in Drosophila. This review summarizes many years of research on FXD in Drosophila models.

Original languageEnglish (US)
Article number87
Issue number1
StatePublished - Jan 2023


  • Drosophila melanogaster
  • FMR1 gene
  • FMRP
  • Fragile X syndrome

ASJC Scopus subject areas

  • Genetics(clinical)
  • Genetics


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