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Background: Action observation treatment (AOT) is an innovative therapeutic approach consisting in the observation of actions followed by their subsequent repetition. The standard version of AOT consists in the observation/imitation of a typically developed individual, which is proposed as model (TDM-AOT)., Aim: This study aims to compare the effectiveness of AOT based on a pathological ameliorative model (PAM-AOT) versus TDM-AOT in improving upper limb ability in children with unilateral cerebral palsy (UCP)., Design: The study consists in a prospective randomized controlled, evaluator-blinded trial (RCT), with two active arms, designed to evaluate the effectiveness of AOT based on pathological model (PAM-AOT) as compared to a standard AOT based on TDM (TDM-AOT)., Setting: The 3-week AOT program was administered in a clinical setting. For some patients, the treatment was delivered at participant's home with the remote support of the physiotherapist (tele-rehabilitation)., Population: Twenty-six children with UCP (mean age 10.5±3.09 years; 14 females) participated in the study, with the experimental group observing a pathological model and the control group observing a typically developed model., Methods: Motor assessments included unimanual and bimanual ability measures conducted at T0 (baseline, before the treatment), T1 (3 weeks after T0), T2 (8-12 weeks after treatment) and T3 (24-28 weeks after treatment); a subset of 16 patients also underwent fMRI motor assessment. Generalized Estimating Equations models were used for statistical analysis., Results: Both groups showed significant improvement in bimanual function (GEE, Wald 106.16; P<0.001) at T1 (P<0.001), T2 (P<0.001), and T3 (P<0.001). Noteworthy, the experimental group showed greater improvement than the control group immediately after treatment (P<0.013). Both groups exhibited similar improvement in unimanual ability (GEE, Wald 25.49; P<0.001). The fMRI assessments revealed increased activation of ventral premotor cortex after treatment in the experimental compared with control group (GEE, Wald 6.26; P<0.012)., Conclusions: Overall, this study highlights the effectiveness of PAM-AOT in achieving short-term improvement of upper limb ability in children with UCP., Clinical Rehabilitation Impact: These findings have significant implications for rehabilitative interventions based on AOT in hemiplegic children, by proposing a non-traditional approach focused on the most functional improvement achievable by imitating a pathological model.