Background <p>Caregiver burden in Frontotemporal Dementia (FTD) is greater than in other forms of dementia. Most of the research has focused on spouses’ burden but the impact on offspring has not been investigated to date, even though FTD is often a presenile dementia.</p> Objective <p>To evaluate the burden of offspring caregivers (OC) of patients with FTD.</p> Methods <p>A total of 55 OC of patients with FTD were included and interviewed. OC burden was</p> <p>assessed by Burden Assessment Scale (BAS). Predictors of OC burden were assessed, considering <i>a)</i> OC demographic features, and <i>b)</i> primary caregiver (PC) (i.e., spouses) demographic features and burden, and <i>c)</i> FTD patient’ related-features.</p> Results <p>OC sex was the only significant predictor of BAS scores (<i>p</i> = 0.011), with females offspring reporting higher burden compared to males. Females OC of more educated PC perceive greater burden (<i>p</i> = 0.004), while younger age in males OC was associated with greater burden (<i>p</i> =0.016). In cohabitants, OC burden was significantly associated with PC burden (<i>p</i> &lt; 0.001).</p> Conclusions <p>The data suggest that supportive programs should be developed not only for PC but also for OC of patients with FTD, especially in females and in cohabitants OC.</p>