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To test the hypothesis that genetic mutations causing frontotemporal dementia have neurodevelopmental consequences, we examined the youngest adults in the GENFI cohort of pre-symptomatic frontotemporal dementia mutation carriers who are between 19 and 30 years of age.<\/jats:p><jats:p>Structural brain differences and improved performance on some cognitive tests were found for MAPT and GRN mutation carriers relative to familial non-carriers, while smaller volumes were observed in C9orf72 repeat expansion carriers at a mean age of 26 years.<\/jats:p><jats:p>The detection of such early differences supports potential advantageous neurodevelopmental consequences of some frontotemporal dementia-causing genetic mutations. These results have implications for the design of therapeutic interventions for frontotemporal dementia. Future studies at younger ages are needed to identify specific early pathophysiologic or compensatory processes that occur during the neurodevelopmental period.<\/jats:p>","DOI":"10.1093\/brain\/awac446","type":"journal-article","created":{"date-parts":[[2022,11,30]],"date-time":"2022-11-30T17:14:12Z","timestamp":1669828452000},"page":"2120-2131","source":"Crossref","is-referenced-by-count":25,"title":["Neurodevelopmental effects of genetic frontotemporal dementia in young adult mutation carriers"],"prefix":"10.1093","volume":"146","author":[{"given":"Elizabeth","family":"Finger","sequence":"first","affiliation":[{"name":"Department of Clinical Neurological Sciences, University of Western Ontario , London, ON , Canada"}]},{"given":"Rubina","family":"Malik","sequence":"additional","affiliation":[{"name":"Schulich School of Medicine & Dentistry, Graduate Program in Neuroscience, University of Western Ontario , London, Ontario , Canada"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1814-5024","authenticated-orcid":false,"given":"Martina","family":"Bocchetta","sequence":"additional","affiliation":[{"name":"Dementia Research Centre, Department of Neurodegenerative Disease, UCL Queen Square Institute of Neurology, University College London , London , UK"},{"name":"Centre for Cognitive and Clinical Neuroscience, Division of Psychology, Department of Life Sciences, College of Health, Medicine and Life Sciences, Brunel University London , London , UK"}]},{"given":"Kristy","family":"Coleman","sequence":"additional","affiliation":[{"name":"Department of Clinical Neurological Sciences, University of Western Ontario , London, ON , Canada"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9949-2951","authenticated-orcid":false,"given":"Caroline","family":"Graff","sequence":"additional","affiliation":[{"name":"Division of Neurogeriatrics, Department NVS, Karolinska Institutet , Stockholm , Sweden"},{"name":"Unit for Hereditary Dementia, Theme Aging, Karolinska University Hospital-Solna , Stockholm , Sweden"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9340-9814","authenticated-orcid":false,"given":"Barbara","family":"Borroni","sequence":"additional","affiliation":[{"name":"Centre for Neurodegenerative Disorders, Neurology Unit, Department of Clinical and Experimental Sciences, University of Brescia , Brescia , Italy"}]},{"given":"Mario","family":"Masellis","sequence":"additional","affiliation":[{"name":"L.C. 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