J Korean Assoc Oral Maxillofac Surg.  2019 Jun;45(3):167-172. 10.5125/jkaoms.2019.45.3.167.

Langerhans cell histiocytosis of the mandible: two case reports and literature review

• Hwang DS
• Lee JS
• Kim UK
• Park HR
• Ryu MH
• Lee JH
• Jung YH
• Kim GC

• KMID: 2451324
• DOI: http://doi.org/10.5125/jkaoms.2019.45.3.167

Abstract

Langerhans cell histiocytosis (LCH) is a rare disorder characterized by the proliferation of dendritic cells resulting in local or systemic symptoms. The clinical symptoms of patients with Langerhans cell histiocytosis depend on the site and the degree of involvement. This article describes two case histories of unifocal bony Langerhans cell histiocytosis with mandibular involvement and further discusses the appropriate management of such via a review of the literature.

Keyword

Langerhans cell histiocytosis; Unifocal Langerhans cell histiocytosis

MeSH Terms

Dendritic Cells
Histiocytosis, Langerhans-Cell*
Humans
Mandible*

Figure

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  Fig. 1 Clinical and radiological feature. A. Intraoral photograph. Despite the presence of right mandibular swelling, there was no specific finding in the intraoral findings. B. Panoramic view. It was difficult to locate specific findings on panoramic examination. C, D. Osteolytic findings were observed on computed tomography (CT), including a well-defined, buccolingual expansile lesion (C: axial view, D: coronal view). Profuse periosteal reaction was also observed around the lesion.

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  Fig. 2 Histologic feature. Langerhans cells and eosinophils are densely infiltrated. It was proved to be Langerhans cell histiocytosis (CD1a/S-100 positive) on additional immunohistological staining (H&E staining, ×200).

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  Fig. 3 Bone scan images. A. Before therapy. A focal uptake was observed in the right mandible. B. After therapy. No abnormal uptake was observed.

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  Fig. 4 Clinical and radiological feature. A. Intraoral photograph. Clinical examination revealed swelling of the right mandible, pain in the affected area, mobility of #85, and gingival swelling and bleeding. B. Panoramic view. It was difficult to locate specific findings on panoramic examination. C, D. Osteolytic findings were observed on computed tomography (CT), including a well-defined, buccolingual expansile lesion (C: axial view, D: coronal view). Profuse periosteal reaction was also observed around the lesion.

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  Fig. 5 Histologic feature. Langerhans cells and eosinophils are densely infiltrated. It was proved to be Langerhans cell histiocytosis (CD1a/S-100 positive) on additional immunohistological staining (H&E staining, ×200).

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  Fig. 6 Bone scan images. A. Before therapy. A focal uptake was observed in the right mandible. B. After therapy. No abnormal uptake was observed.

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