Mandibular single-system Langerhans cell histiocytosis with repeated recurrences at other sites, and a brief literature review

Abstract

Langerhans cell histiocytosis (LCH) is rare disease that has a very diverse course, ranging from spontaneous recovery to fatal disease. We report the case of a 2-year-old Japanese boy who visited our department with the chief complaint of a mass formation in the molar region of his right mandible. Odontogenic infection was suspected for the mass, and the patient had been receiving antibiotics at a nearby pediatrician’s clinic for about 1 month before visiting our department, but the lesion had increased in size. The patient was admitted to the hospital and an excisional biopsy was performed with general anesthesia. Based on the pathological test results, we diagnosed mandibular single-system LCH. Chemotherapy was performed our hospital’s pediatric department. Thereafter, recurrence occurred four times in the mandible and skull, and chemotherapy was administered each time. Over the 4.5 years since the initial diagnosis, there have been no further signs of recurrence or any effects on the growth of the patient’s jawbone or eruption of teeth.