Premature infants are at risk for cerebral palsy (CP) that is typically diagnosed between 18–24 months. We present a case study of an infant who was discharged from the neonatal intensive care unit (NICU) without obvious neurological deficits but was later diagnosed with hemiplegic CP. The infant was enrolled in an infant motor study, which included neuroimaging and developmental motor assessments. At term, anatomical MRI showed bilateral periventricular leukomalacia, abnormal brain metabolites in frontal white matter via MR spectroscopy (MRS), and low fractional anisotropy (FA) values obtained from diffusional kurtosis imaging (DKI) in several cortical white matter tracts compared to a group of typically developing infants without neuroimaging abnormalities. In addition, the infant scored below average on a developmental assessment administered at term and three months as well as on the standard Bayley III assessment at 12 months. Abnormal neuroimaging and low scores on the early developmental assessment prompted referral for intervention services at two months. With intensive therapy, by 45 months, the infant was average in self-care, mobility, and communication skills, although below average in visual motor and gross motor coordination. This case highlights the clinical impact of early detection and referral using combined neuroimaging and developmental testing.