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Description: Why do we sing to babies? Human infants are relatively altricial and need their parents' attention to survive. Infant-directed song may constitute a signal of that attention. In the rare genomic imprinting disorder Prader-Willi syndrome (PWS), typically paternally-expressed genes from chromosome 15q11–q13 are unexpressed, resulting in exaggeration of traits that reduce offspring investment demands on the mother. PWS may thus be associated with a distinctive musical phenotype. We report unusual responses to music in PWS. Subjects (N = 39) moved more during music listening, exhibited greater reductions in heart rate in response to music listening, and displayed a specific deficit in pitch discrimination ability relative to typically-developing adults and children (N = 589). Paternally-expressed genes from 15q11–q13, unexpressed in PWS, may thus increase demands for music and enhance perceptual sensitivity to music. These results implicate genomic imprinting in the psychology of music, informing theories of music's evolutionary history.