The sensory input that we experience is highly patterned, and we are experts at detecting these regularities. Although the extraction of such regularities, or statistical learning (SL), is typically viewed as a cortical process, recent studies have implicated the medial temporal lobe (MTL), including the hippocampus. These studies have employed fMRI, leaving open the possibility that the MTL is involved but not necessary for SL. Here, we examined this issue in a case study of LSJ, a patient with complete bilateral hippocampal loss and broader MTL damage. In Experiments 1 and 2, LSJ and matched control participants were passively exposed to a continuous sequence of shapes, syllables, scenes, or tones containing temporal regularities in the co-occurrence of items. In a subsequent test phase, the control groups exhibited reliable SL in all conditions, successfully discriminating regularities from recombinations of the same items into novel foil sequences. LSJ, however, exhibited no SL, failing to discriminate regularities from foils. Experiment 3 ruled out more general explanations for this failure, such as inattention during exposure or difficulty following test instructions, by showing that LSJ could discriminate which individual items had been exposed. These findings provide converging support for the importance of the MTL in extracting temporal regularities.