Stroke in young adults is considered a rare pathology, associated with a significant morbidity and mortality rate. In the etiological study of stroke in young people, it is important to carry out a comprehensive investigation as there is a greater propensity for undetermined causes, compared to the older population. Here, the authors present the clinical case of a 53-year-old male diagnosed with ischemic stroke, in which an extensive evaluation identified left ventricular hypertrabeculation as the presumed etiology of this event. This finding highlights the extreme importance of the etiological study, being particularly interesting due to the rarity of the findings.