Background: Immunological dysfunction is known to be present in Langerhans cell histiocytosis; some autoimmune diseases with autoantibodies associated with this condition have already been described. This immunological dysfunction could play a role in the Langerhans cell histiocytosis pathogenesis.
Case report: A 13 year-old boy presented a multifocal bone Langerhans cell histiocytosis associated with Graves' syndrome. This last condition was successfully treated with carbimazole while the focal lesions of histiocytosis did not require any treatment.
Conclusion: This unique association could be explained by the immunological dysfunction seen in Langerhans cell histiocytosis with secondary appearance of thyreo-stimulating antibodies.