Jan Rustemeyer1, Birte Julia Siegmund2, Yunus Okcu2, Alexander Busch2. 1. Department of Oral and Maxillofacial Surgery and Plastic Operations, Klinikum Bremen-Mitte, School of Medicine, University of Göttingen, Bremen, Germany. janrustem@gmx.de. 2. Department of Oral and Maxillofacial Surgery and Plastic Operations, Klinikum Bremen-Mitte, School of Medicine, University of Göttingen, Bremen, Germany.
Abstract
BACKGROUND: Diffuse sclerosing osteomyelitis (DSO) is a non-purulent chronic recurrent inflammation and affects the mandible in many cases. Belonging to the group of autoinflammatory diseases, in children and in cases with various additional symptoms including synovitis, acne, pustulosis, hyerostosis, and osteitis (SAPHO syndrome), therapy usually consists of non-surgical treatment. Against this background, we present an unusual course of DSO in an adult female patient. CASE REPORT: A 50-year-old female suffering from DSO without SAPHO syndrome was pretreated for years with conservative drug regimens and local surgery. Previous therapy was not successful, and subsequently, multiple surgical procedures were carried out focused on recurrent acute exacerbations of DSO. Surgery resulted in a total resection and alloplastic and autoplastic reconstruction of the mandible including both temporomandibular joints. Prosthetic rehabilitation was possible after dental implant loading, and the final outcome was very satisfactory. CONCLUSION: In the event that non-surgical options are not successful in DSO, an extended surgical therapy becomes necessary. Even if surgery results in complete resection of the mandible, a satisfactory rehabilitation can be achieved after complex reconstruction.
BACKGROUND: Diffuse sclerosing osteomyelitis (DSO) is a non-purulent chronic recurrent inflammation and affects the mandible in many cases. Belonging to the group of autoinflammatory diseases, in children and in cases with various additional symptoms including synovitis, acne, pustulosis, hyerostosis, and osteitis (SAPHO syndrome), therapy usually consists of non-surgical treatment. Against this background, we present an unusual course of DSO in an adult female patient. CASE REPORT: A 50-year-old female suffering from DSO without SAPHO syndrome was pretreated for years with conservative drug regimens and local surgery. Previous therapy was not successful, and subsequently, multiple surgical procedures were carried out focused on recurrent acute exacerbations of DSO. Surgery resulted in a total resection and alloplastic and autoplastic reconstruction of the mandible including both temporomandibular joints. Prosthetic rehabilitation was possible after dental implant loading, and the final outcome was very satisfactory. CONCLUSION: In the event that non-surgical options are not successful in DSO, an extended surgical therapy becomes necessary. Even if surgery results in complete resection of the mandible, a satisfactory rehabilitation can be achieved after complex reconstruction.
Authors: Marieke M van de Meent; Hamid Meshkini; Marta Fiocco; Miranda J M Wetselaar-Glas; Natasha M Appelman-Dijkstra; J P Richard van Merkesteyn Journal: J Craniomaxillofac Surg Date: 2017-10-03 Impact factor: 2.078