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.
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References
Kahn MF, Hayem F, Hayem G, Grossin M (1994) Is diffuse sclerosing osteomyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome? Analysis of seven cases. Oral Surg Oral Med Oral Pathol 78:594–598
Roldán JC, Terheyden H, Dunsche A, Kampen WU, Schroeder JO (2001) Acne with chronic recurrent multifocal osteomyelitis involving the mandible as part of the SAPHO syndrome: case report. Br J Oral Maxillofac Surg 39:141–144
Girschick HJ, Zimmer C, Klaus G, Darge K, Dick A, Morbach H (2007) Chronic recurrent multifocal osteomyelitis: what is it and how should it be treated? Nat Clin Pract Rheumatol 3:733–738
Hofmann SR, Kapplusch F, Girschick HJ, Morbach H, Pablik J, Ferguson PJ, Hedrich CM (2017) Chronic recurrent multifocal osteomyelitis (CRMO): presentation, pathogenesis, and treatment. Curr Osteoporos Rep 15:542–554
van de Meent MM, Meshkini H, Fiocco M, Wetselaar-Glas MJM, Appelman-Dijkstra NM, van Merkesteyn JPR (2017) Conservative treatment of children with chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible. J Craniomaxillofac Surg 45:1938–1943
Sugata T, Fujita Y, Myoken Y, Kiriyama T (2003) Successful management of severe facial pain in patients with diffuse sclerosing osteomyelitis (DSO) of the mandible using disodium clodronate. Int J Oral Maxillofac Surg 32:574–575
Senel FC, Saracoglu Tekin U, Durmus A, Bagis B (2007) Severe osteomyelitis of the mandible associated with the use of non-nitrogen-containing bisphosphonate (disodium clodronate): report of a case. J Oral Maxillofac Surg 65:562–565
Otto S, Troeltzsch M, Burian E, Mahaini S, Probst FA, Pautke C, Ehrenfeld M, Smolka W (2015) Ibandronate treatment of diffuse sclerosing osteomyelitis of the mandible: pain relief and insight into pathogenesis. J Craniomaxillofac Surg 43:1837–1842
Otto S, Burian E, Troeltzsch M, Kaeppler G, Ehrenfeld M (2018) Denosumab as a potential treatment alternative for patients suffering from diffuse sclerosing osteomyelitis of the mandible - rapid communication. J Craniomaxillofac Surg 46:534–537
Balaji SM (2016) Total reconstruction of mandible by transport distraction after complete resection for benign and malignant tumors. Indian J Dent Res 27:205–212
Abe T, Kitada H, Sato J, Yamazaki Y, Notani K, Yamaguchi T, Kitagawa Y (2011) Successful total mandibular reconstruction using a fibular graft with long-term follow-up: a case report. Asian J Oral Maxillofac Surg 23:196–200
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Rustemeyer, J., Siegmund, B.J., Okcu, Y. et al. Total mandibular reconstruction following diffuse sclerosing osteomyelitis. Oral Maxillofac Surg 23, 95–99 (2019). https://doi.org/10.1007/s10006-018-0731-9
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DOI: https://doi.org/10.1007/s10006-018-0731-9