Abstract

We report two patients with Cole-Engman syndrome (dyskeratosis congenita) .
Case 1: A 48-year-old Japanese man had noticed reticulated skin hyperpigmentation on his whole body from 11-year-old. He came to our dental hospital with acomplain of a tumor in the oral cavity. It was diagnosed as leukoplakia and squamous cell carcinoma arising from leukoplakia of the mucous menbranes on the left mandibular gingiva. He had also dental anomalies, nail dystrophy and lacrimal duct obstruction. He was diagnosed as Cole-Engman syndrome, comlete type.
Case 2: A 49-year-old Japanese man visited our hospital with a complain of nummular eczema and prurigo-like eruption with itching. He also had reticulated skin hyperpigmentation, nail dyatrophy, hyperkeratosis of palms and soles, angular stomatitis and leukoplakia on right lateral side of tongue.
We diagnosed him as Cole-Engman syndrome, comlete type.
We followed up them very carefully.