Histiocytosis X (HX) arising in the oral and maxillofacial region is relatively rare. Here, two cases of HX, i.e., Hand-Schaller-Christian disease (HSC) and Eosinophilic granuloma of bone which occurred in the oral and maxillofacial region are presented with immunohistochemical, light and electronmicroscopic studies.
Case 1: a 37-year-old male was referred to our hospital, with the chief complaint of difficulty in chewing and radiolucent lesions in the bilateral mandible. Pathological examination revealed Eosinophilic granuloma of bone.
Case 2: a 23-year-old male was admitted to our hospital because of the mobility of the front teeth in the mandible. He had received treatment for HSC since he was 1 year and 6 months old.
Electronmicroscopic observations showed a number of proliferated histiocytic cells in both cases and these cells seemed to be Langerhans cells.
In the HSC of which the clinical activity seemed to be higher than that of Eosinophilic granuloma, a number of small Birbeck granules were found as compared with in the Eosinophilic granuloma.
