J Korean Dent Sci.  2016 Jun;9(1):35-41. 10.5856/JKDS.2016.9.1.35.

Treatment and Rehabilitation of Repetitively Recurrent Langerhans Cell Histiocytosis: A Case Report

• Yoo HY
• Park KS
• Lee BS
• Kwon YD
• Choi BJ
• Ohe JY
• Lee JW

• KMID: 2382976
• DOI: http://doi.org/10.5856/JKDS.2016.9.1.35

Abstract

Langerhans cell histiocytosis (LCH) is characterized by proliferation of histiocyte-like cells (Langerhans cell histiocytes) with characteristic Birbeck granules, accompanied by other inflammatory cells. Treatments of LCH include surgery, chemotherapy, and radiotherapy. One of the representative forms of chemotherapy is intralesional injection of steroids. Surgical treatment in the form of simple excision, curettage, or even ostectomy can be performed depending on the extent of involvement. Radiotherapy is suggested in case of local recurrence, or a widespread lesion. This article shows the case of repetitively recurrent LCH of a 56-year-old man who had been through surgical excision and had to have marginal mandibulectomy and radiotherapy when the disease recurred. After the first recurrence occurred, lesions involved the extensive part of the mandible causing pathologic fracture, so partial mandibular bone resection was performed from the right molar area to the left molar area followed by the excision of the surrounding infected soft tissues. The resected mandibular bone was reconstructed with a segment of fibula osteomyocutaneous free flap and overdenture prosthesis supported by osseointegrated implants.

Keyword

Fibula osteomyocutaneous free flap; Histiocytosis, Langerhans cell; Reconstruction; Recurrence

MeSH Terms

Curettage
Denture, Overlay
Drug Therapy
Fibula
Fractures, Spontaneous
Free Tissue Flaps
Histiocytosis, Langerhans-Cell*
Humans
Injections, Intralesional
Mandible
Middle Aged
Molar
Prostheses and Implants
Radiotherapy
Recurrence
Rehabilitation*
Steroids
Steroids