Although the characteristic clinical appearance and the differences in distribution, it is often difficult to differentiate keratosis follicularis squamosa (Dohi) from other keratotic disorders. Here, we describe the case of a 5-year-old boy with Dohi in the first time that by using dermoscopy. Dermoscpoy of the lesion showed typical lotus leaves on the water appearance and follicular plug in the center. We reported the usefulness of dermosocopy in Dohi.