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Int J Clin Exp Pathol 2013;6(5):939-942

Case Report
Recurrent multifocal Langerhans cell histiocytosis of the mandible and maxilla in a
46-year-old man: a pathologic case report

Tadashi Terada

Department of Pathology, Shizuoka City Shimizu Hospital, Shizuoka, Japan

Received September 24, 2012; Accepted March 20, 2013; Epub April 15, 2013; Published May 1, 2013

Abstract: A 46-year-old man consulted to a private dental clinic for tooth extraction, where he was indicated to have abnormal shadow in the
right mandible. The patient was referred to our clinic hospital. X-ray examination revealed an osteolytic lesion (3 x 2 x 1 cm), and tumor excision
was performed. Pathological diagnosis was difficult. The tumor consisted of round cells with moderate atypia. Nuclear grooves were
recognized. Immunohistochemistry showed positive CD1a and S100 protein. The Ki67 labeling was 16%. The author diagnosed the lesion as
Langerhans cell histiocytosis (LCH). The patient became free of tumor, and discharged. However, the tumor recurred 5 years later. Two
osteolytic lesions were found: one is mandible (3 x 1 x 1 cm), and another was maxilla (0.5 x 0.5 x 0.4 cm). Tumorectomy with wide margins
were performed. The pathological diagnosis was LCH in both lesions. Whole body CT, MRI and PET were performed, but revealed no tumors.
The patient is now free from tumor, and is followed up 7 years after the first presentation. (IJCEP1209027).

Keywords: Langerhans cell histiocytosis (LCH), mandible, pathology, therapy

Address correspondence to: Dr. Tadashi Terada, Department of Pathology, Shizuoka City Shimizu Hospital, Miyakami 1231 Shimizu-Ku,
Shizuoka 424-8636, Japan. Phone: 81-54-336-1111; Fax: 81-54-336-1315; E-mail: piyo0111jp@yahoo.co.jp