Abstract: Background: There are relatively few reports on the histopathological characteristics of diffuse sclerosing osteomyelitis of the mandible (DSOM), which is difficult to distinguish from chronic suppurative osteomyelitis (CSO) and craniofacial fibrous dysplasia (CFD). This study aimed to summarize and compare the histopathological characteristics of DSOM, CFD, and CSO.
Materials and Methods: In this study, hematoxylin and eosin-stained sections of patients with DSOM, CSO, and CFD at the Peking University Hospital of Stomatology from 2015 to 2020 were retrieved. The histopathological characteristics were summarized, including new bone formation, inflammatory cell infiltration, bone trabecular morphology, osteoclasts, sequestrum, bacterial mass, and calcified spherules, similar to cementicles. The histopathological characteristics of DSOM, CSO, and CFD were compared, and the results were statistically analyzed.
Results: In total, 50, 13, and 10 patients with DSOM, CSO, and CFD were included in this study, respectively. In terms of new bone formation, both DSOM and CSO showed reactive bone formation (p = 1), whereas CFD mainly showed fiber osteogenesis (p < 0.001). The inflammatory cells of DSOM were mainly lymphocytes and plasma cells, whereas those of CSO were mainly lymphocytes and neutrophils (p < 0.001), and there was usually no inflammatory cell infiltration in the CFD specimens (p < 0.001). DSOM, CSO, and CFD showed irregular bone trabeculae (p = 0.045, p = 0.703) and active osteoclasts (p1 = 0.189, p2 = 0.256). DSOM showed a small amount of bacterial mass but no sequestrum; neither of which was found in CFD (p = 1, p = 1), but it was common in CSO (p = 0.011 and p = 0.025). DSOM and CSO showed smooth and regular basophilic lines (p = 0.308), whereas CFD showed a rough and irregular basophilic line (p < 0.001).
Conclusions: The histopathological characteristics of the three diseases were partly similar, but there were evident differences. The main differences are the type of new bone formation, types and distribution of inflammatory cells, and presence of sequestrum and bacterial masses. These differences will help clinicians diagnose DSOM.
(© 2022 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.)
References: Marzola C. Chronic diffuse sclerosing osteomyelitis of the maxilla and the mandible. Rev Bras Odontol. 1970;27(165):238-241.
Jacobsson S. Diffuse sclerosing osteomyelitis of the mandible. Int J Oral Surg. 1984;13(5):363-385.
Renapurkar S, Pasternack MS, Nielsen GP, Kaban LB. Juvenile mandibular chronic osteomyelitis: role of surgical debridement and antibiotics. J Oral Maxillofac Surg. 2016;74(7):1368-1382.
Obel G, Krogdahl A, Thygesen T, Godballe C. Juvenile mandibular chronic osteomyelitis: 3 cases and a literature review. J Oral Maxillofac Surg. 2013;71(2):305-309.
van de Meent MM, Pichardo SEC, Rodrigues MF, Verbist BM, van Merkesteyn JPR. Radiographic characteristics of chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible: a comparison with chronic suppurative osteomyelitis and osteoradionecrosis. J Craniomaxillofac Surg. 2018;46(9):1631-1636.
Jia K, Li X, An J, Zhang Y. Comparing clinical and radiographic characteristics of chronic diffuse sclerosing osteomyelitis and craniofacial fibrous dysplasia in the mandible. J Oral Maxillofac Surg. 2021;79(5):1053-1061.
Mallikarjun K, Kohli A, Kumar A, Tanwar A. Chronic suppurative osteomyelitis of the mandible. J Indian Soc Pedod Prev Dent. 2011;29(2):176-179.
Horst SA, Hoerr V, Beineke A, et al. A novel mouse model of Staphylococcus aureus chronic osteomyelitis that closely mimics the human infection: an integrated view of disease pathogenesis. Am J Pathol. 2012;181(4):1206-1214.
Yang L, Wu H, Lu J, Teng L. Prevalence of different forms and involved bones of craniofacial fibrous dysplasia. J Craniofac Surg. 2017;28(1):21-25.
Burke AB, Collins MT, Boyce AM. Fibrous dysplasia of bone: craniofacial and dental implications. Oral Dis. 2017;23(6):697-708.
Assaf AT, Benecke AW, Riecke B, et al. Craniofacial fibrous dysplasia (CFD) of the maxilla in an 11-year old boy: a case report. J Craniomaxillofac Surg. 2012;40(8):788-792.
Van Merkesteyn JP, Groot RH, Bras J, Bakker DJ. Diffuse sclerosing osteomyelitis of the mandible: clinical radiographic and histologic findings in twenty-seven patients. J Oral Maxillofac Surg. 1988;46(10):825-829.
Montonen M, Li TF, Lukinmaa PL, et al. RANKL and cathepsin K in diffuse sclerosing osteomyelitis of the mandible. J Oral Pathol Med. 2006;35(10):620-625.
Jeyaraj P. Histological diversity, diagnostic challenges, and surgical treatment strategies of fibrous dysplasia of upper and mid-thirds of the craniomaxillofacial complex. Ann Maxillofac Surg. 2019;9(2):289-314.
Pereira T, Gomes CC, Brennan PA, Fonseca FP, Gomez RS. Fibrous dysplasia of the jaws: integrating molecular pathogenesis with clinical, radiological, and histopathological features. J Oral Pathol Med. 2019;48(1):3-9.
El-Mofty SK, Nelson B, Toyosawa S. Fibrous dysplasia. WHO Classification of Head and Neck Tumors. 4th ed. IARC; 2017:253-254.
Hofmann SR, Schwarz T, Moller JC, et al. Chronic non-bacterial osteomyelitis is associated with impaired Sp1 signaling, reduced IL10 promoter phosphorylation, and reduced myeloid IL-10 expression. Clin Immunol. 2011;141(3):317-327.
Wagner AD, Andresen J, Jendro MC, Hulsemann JL, Zeidler H. Sustained response to tumor necrosis factor alpha-blocking agents in two patients with SAPHO syndrome. Arthritis Rheum. 2002;46(7):1965-1968.
Jansson A, Renner ED, Ramser J, et al. Classification of non-bacterial osteitis: retrospective study of clinical, immunological and genetic aspects in 89 patients. Rheumatology. 2007;46(1):154-160.
Hofmann SR, Morbach H, Schwarz T, Rösen-Wolff A, Girschick HJ, Hedrich CM. Attenuated TLR4/MAPK signaling in monocytes from patients with CRMO results in impaired IL-10 expression. Clin Immunol. 2012;145(1):69-76.
Hofmann SR, Kubasch AS, Ioannidis C, et al. Altered expression of IL-10 family cytokines in monocytes from CRMO patients result in enhanced IL-1beta expression and release. Clin Immunol. 2015;161(2):300-307.
McDermott MF, Aksentijevich I, Galon J, et al. Germline mutations in the extracellular domains of the 55 kDa TNF receptor, TNFR1, define a family of dominantly inherited autoinflammatory syndromes. Cell. 1999;97(1):133-144.
McDermott MF. A common pathway in periodic fever syndromes. Trends Immunol. 2004;25(9):457-460.
Malmstrom M, Fyhrquist F, Kosunen TU, Tasanen A. Immunological features of patients with chronic sclerosing osteomyelitis of the mandible. Int J Oral Surg. 1983;12(1):6-13.
Chitu V, Ferguson PJ, de Bruijn R, et al. Primed innate immunity leads to autoinflammatory disease in PSTPIP2-deficient cmo mice. Blood. 2009;114(12):2497-2505.
Idowu BD, Al-Adnani M, O'Donnell P, et al. A sensitive mutation-specific screening technique for GNAS1 mutations in cases of fibrous dysplasia: the first report of a codon 227 mutation in bone. Histopathology. 2007;50(6):691-704.
Weinstein LS, Liu J, Sakamoto A, Xie T, Chen M. Minireview: GNAS: normal and abnormal functions. Endocrinology. 2004;145(12):5459-5464.
Godse AS, Shrotriya SP, Vaid NS. Fibrous dysplasia of the maxilla. J Pediatr Surg. 2009;44(4):849-851.
Sybenga AB, Jupiter DC, Speights VO, Rao A. Diagnosing osteomyelitis: a histology guide for pathologists. J Foot Ankle Surg. 2020;59(1):75-85.
Jacobsson S, Dahlen G, Moller AJ. Bacteriologic and serologic investigation in diffuse sclerosing osteomyelitis (DSO) of the mandible. Oral Surg Oral Med Oral Pathol. 1982;54(5):506-512.
Jacobsson S, Hallen O, Hollender L, Hansson CG, Lindstrom J. Fibro-osseous lesion of the mandible mimicking chronic osteomyelitis. Oral Surg Oral Med Oral Pathol. 1975;40(4):433-444.
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