Intralesional corticosteroid therapy for mandibular Langerhans cell histiocytosis preserving the intralesional tooth germ.
BACKGROUND
Langerhans cell histiocytosis (LCH, histiocytosis X, ICD-O 9751/1) refers to a neoplastic proliferation of Langerhans cells. The course of the disease determines the treatment and prognosis. Solitary forms (eosinophilic granuloma) and limited multilocational lesions may be treated successfully with local surgical intervention and intralesional corticosteroid injection.


OBJECTIVE
Presentation of our own case will review LCH, a very rare disease entity in oral-facial surgery and will document that intralesional corticosteroid injection is a less invasive alternative to the classical surgical curettage and local radiation therapy.


METHODS
In a 10-year-old boy, a progressive, pressure-sensitive swelling had developed within 1 week in the left paramandibular and submandibular area. Dental status was good. Diagnostic imaging demonstrated a diffusely contoured osteolysis caudal to tooth germ 37 with infiltration of the adjacent chewing muscles. Histological evaluation of the intraoral biopsy sample established an LCH. Having excluded a multifocal form of LCH, treatment with intralesional injection of methylprednisolone 200 mg was chosen. Symptoms of pain were quickly relieved and the swelling receded. Follow-up visits 6 weeks, 3 months, and 6 months after corticosteroid injection revealed continuous regression of mandibular osteolysis. OPG and MRI after 17 months demonstrated a good osseous consolidation in the left mandibular angle area, but a still discernible bone marrow edema. Development of the intralesional tooth germ 37 appeared normal.


CONCLUSIONS
Local surgical interventions continue to be central to the range of accepted therapeutic measures. However, the increasing numbers of reports on the successful treatment of solitary LCH by intralesional corticosteroid injection suggest that this treatment option should be considered especially in children to preserve tooth germs.
Moralis
Antonios
A
Klinik und Poliklinik für Mund-, Kiefer- und Gesichtschirurgie, Universität Regensburg, Franz-Josef-Strauss-Allee 11, Regensburg, Germany.
Kunkel
Martin
M
Kleinsasser
Norbert
N
Müller-Richter
Urs
U
Reichert
Torsten Eugen
TE
Driemel
Oliver
O
D000893:Anti-Inflammatory Agents / Q000008:administration & dosage; D002648:Child; D005500:Follow-Up Studies; D005938:Glucocorticoids / Q000008:administration & dosage*; D006646:Histiocytosis, Langerhans-Cell / Q000188:drug therapy* / Q000473:pathology; D006801:Humans; D036502:Infusions, Intralesional; D008297:Male; D008336:Mandibular Diseases / Q000188:drug therapy* / Q000473:pathology; D008339:Mandibular Neoplasms / Q000473:pathology; D008775:Methylprednisolone / Q000008:administration & dosage*; D014083:Tooth Germ / Q000187:drug effects / Q000473:pathology; D016896:Treatment Outcome
D000893:Anti-Inflammatory Agents; D005938:Glucocorticoids; D008775:Methylprednisolone