An overlooked case of mandibular histiocytosis: Case report and review of the literature

Abstract

Introduction: Langerhans histiocytosis is a rare disorder resulting from the proliferation and accumulation of a specific histiocytic cell called the Langerhans cell. Mandibular histiocytosis specifically can be asymptomatic or manifest as a jaw mass, Orthopanthogram as well as CT are required for the early diagnosis and prevention of total destruction of the osseous bone. \nCase Report: We report the case of a 7-years-old patient, with a history of a non-resolving right jaw swelling treated as a dental abscess with antibiotics and analgesics for 14 days. The patient has no history of trauma, and was referred to our department for a right jaw swelling evolving for three months. Oral examination found an ulceroproliferative process of the posterior aspect of the dental part of the right mandible. Dental contrast enhanced CT showed a destruction of the periodontium, with an extensive geographical osteolytic homogeneous mass destroying the mandible. The diagnosis of histiocytosis was suggested histologically after biopsy. After a 6 weeks regimen of vinblastine and prednisolone with a clear regression, a surgical curettage of the bone lesion was performed. Local control was achieved with no complications in the six months follow up examination. \nConclusion: In conclusion, our case suggests that rapid diagnosis followed by minimally invasive surgery can be determinant in the prevention of possible deformation, especially in the pediatric population. Further investigation of the role of microbiopsy for screening should be discussed. Computed tomography is the first choice imaging technique in the diagnosis of mandibular histiocytosis.