A pathologic mandibular fracture revealing a bifocal location of Langerhans cell histiocytosis

• Published in: Annals of medicine and surgery (2012) Vol. 56; pp. 128 - 132
• Main Authors: Sabrine, Maamouri, Marouen, Ben Rejeb, Riahi, Ines, Karima, Zitouni, Nadia, Zanidi, Issam, Zairi
• Format: Report
• Language: English
• Published: 01-08-2020
• ISSN: 2049-0801; 2049-0801
• DOI: 10.1016/j.amsu.2020.06.019

INTRODUCTIONLangerhans cell histiocytosis is a rare disease. When it occurs in the cranium/facial bones, the mandibular location is the most frequent. PRESENTATION OF CASEA 31 years-old man was referred to our department for a mandibular chronic discomfort during an acute exacerbation, spontaneous teeth mobility and an alteration of the dental occlusion revealing a pathologic mandibular fracture.The diagnosis was confirmed by a subsequent CT scan.The surgical procedure was performed under general anesthesia by a maxillo-facial senior surgeon. The therapeutic plan combined teeth extractions, enucleation of both the left maxillary and right mandibular cystic lesions and osteosynthesis of the pathologic mandibular fracture with a miniplate.Histological and immunohistochemistry analysis of the maxillary and mandibular cystic lesions pointed the diagnosis of a bifocal Langerhans cell histiocytosis of the oral cavity.Several investigations were done in order to find another location, showing no abnormalities. DISCUSSIONThis is a case of rare single system LCH at two distinct locations: one at the mandibular bone and the other at the upper left maxilla. Both were uncovered by an acute exacerbation of a chronic discomfort secondary to a mandibular pathologic fracture. This should draw attention to the issues of the diagnosis. CONCLUSIONAn early LCH diagnosis and a multidisciplinary treatment plan allow the improvement of the patient 's prognosis and quality of life.