The distribution of lentigines offers great insight into the multitude of potential-associated genetic or acquired conditions. In this report, we described a unique presentation of lentigines limited to the palms and soles in a healthy individual. Personal and familial history, clinical examination, serological testing, and whole-genome sequencing were all unremarkable. The benign clinical features and absence of associated medical conditions support the likely lentigo simplex with an isolated palmoplantar distribution. To date, no similar distribution has been reported. This case extends the breadth of view into all possible presentations of lentigines.