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Clinical features of behavioural and language dysfunction are associated with neurodegeneration, predominantly of frontal and temporal cortices. Over the past decade, there have been significant advances in the understanding of the genetic aetiology and neuropathology of FTD which have led to the creation of various disease models to investigate the molecular pathways that contribute to disease pathogenesis. The generation of <jats:italic>in\u00a0vivo<\/jats:italic> models of FTD involves either targeting genes with known disease\u2010causative mutations such as <jats:italic>GRN and C9orf72<\/jats:italic> or genes encoding proteins that form the inclusions that characterize the disease pathologically, such as TDP\u201043 and FUS. This review provides a comprehensive summary of the different <jats:italic>in\u00a0vivo<\/jats:italic> model systems used to understand pathomechanisms in FTD, with a focus on disease models which reproduce aspects of the wide\u2010ranging behavioural phenotypes seen in people with FTD. We discuss the emerging disease pathways that have emerged from these <jats:italic>in\u00a0vivo<\/jats:italic> models and how this has shaped our understanding of disease mechanisms underpinning FTD. We also discuss the challenges of modelling the complex clinical symptoms shown by people with FTD, the confounding overlap with features of motor neuron disease, and the drive to make models more disease\u2010relevant. In summary, <jats:italic>in\u00a0vivo<\/jats:italic> models can replicate many pathological and behavioural aspects of clinical FTD, but robust and thorough investigations utilizing shared features and variability between disease models will improve the disease\u2010relevance of findings and thus better inform therapeutic development.<\/jats:p>","DOI":"10.1111\/nan.12536","type":"journal-article","created":{"date-parts":[[2018,12,24]],"date-time":"2018-12-24T05:54:28Z","timestamp":1545630868000},"page":"58-80","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Review: Modelling the pathology and behaviour of frontotemporal dementia"],"prefix":"10.1111","volume":"45","author":[{"given":"D. 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