Reconstruction of a Patient With Rosai-Dorfman Disease Using Ramus Graft and Osseointegrated Implants: A Case Report

Abstract

R osai-Dorfman disease (RDD), or sinus histiocytosis with massive lymphadenopathy, is a rare and benign, frequently self-limited histiocytic disorder of unknown etiology. The most common clinical findings are broad painless lymphadenopathy accompanied by fever, weight loss, tonsillitis, rhinorrhea, nasal obstruction, and sometimes hepatosplenomegaly. Laboratory findings in patients with RDD include an elevated erythrocyte sedimentation rate, anemia, leukocytosis and polyclonal hypergammaglobulinemia. RDD classically presents with painless massive cervical lymphadenopathy, which is caused by accumulation of lymphocytes and histiocytes distending the lymph node sinuses. However, the disease may occur at extranodal sites. In most instances, the extranodal sites include the eyes, head and neck, skin, or bone. Mandibular cortical grafts are the gold standard in the restoration of intraoral osseous defects. These grafts do not produce immune reactions and are incorporated by osteoclastic resorption with a short healing period when compared with other methods of osseous repair. Intraoral donor sites include the mandibular ramus, angle of the mandible, mandibular symphysis, and mandibular exostoses. The mandibular ramus provides primarily a dense cortical graft. Other advantages of a mandibular ramus transplant are minimal resorption, maintenance of osseous density, intraoral access, and proximity to the recipient site. Cortical grafts, including the ramus, are reliable donor sites for the reconstruction of the alveolar ridge deformities before dental implant placement. The highest concentrations of promoter proteins (eg, bone morphogenetic proteins) are found in the mandibular cortical grafts. This report presents a case of an isolated RDD involving the mandible as well as its surgical management, preprosthetic reconstruction, and restoration with dental implants.