Hypoesthesia in the inferior alveolar nerve as the unique clinical manifestation of Rosai-Dorfman disease

Rosai and Dorfman1 first described this clinicopathologic entity in 1969 as a sinus histiocytosis with massive lymphadenopathy (SHML). Although the etiology is still unknown, the disease is thought to be due to a disregulation of the immune system possibly as an abnormal response to an infectious agent. The most frequent clinical manifestation of Rosai-Dorfman disease is bilateral, nontender cervical lymphadenopathy. Extranodal involvement is present in 43% of cases, with preferential affectation of the head and neck region,2 including the upper respiratory tract (73%), orbit (50%), and salivary glands (25%).2,3 It has also been reported in the liver, skin, and central nervous system but not as a peripheral nerve disease.4 The aim of this report was to present a new case of extranodal Rosai-Dorfman disease affecting a peripheral nerve, specifically the mandibular portion of the trigeminal nerve.