Charcot-Leyden crystals (CLCs) have been found in many conditions associated with eosinophilia, but their occurrence in skin diseases is very rare. We report ultrastructural observations on the presence of CLCs in the cutaneous lesions of two cases of mastocytoma. Electron microscopy documented CLCs located in phagosomes of morphologically activated macrophages as well as free CLCs in the stromal tissue, close association between CLCs formation and damaged and lysed eosinophils was present. These findings provided evidence that the formation of CLCs in mastocytoma implicated the individual and interrelated biology of mast cells, eosinophils and macrophages. Phagosomes probably acted as the site of CLCs formation. The clinic and pathologic role of CLCs in mastocytoma deserves further investigation.