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South African Dental Journal
versão On-line ISSN 0375-1562
versão impressa ISSN 0011-8516
S. Afr. dent. j. vol.77 no.4 Johannesburg Mai. 2022
http://dx.doi.org/10.17159/2519-0105/2022/v77no4a9
RADIOLOGY CASE
C SmitI; Z YakoobII
IBChD, MSc (Maxillofacial and Oral Radiology). Department of Oral Pathology and Oral Biology, University of Pretoria. ORCID: 0000-0003-4047-6356
IIBChD, PDD (Maxillofacial and Oral Radiology), MSc (Maxillofacial and Oral Radiology). ORCID: 0000-0003-19665574. Department of Oral Pathology and Oral Biology, University of Pretoria
CASES
Two sisters presented with a family history of chronic episodes of osteomyelitis due to an underlying fibro-osseous bone disorder. The patients reported no co-morbidities or long bone fractures. What are the pertinent radiological features and your diagnostic hypothesis?
INTERPRETATION
Both patients presented with extensive sclerosis limited to the tooth-bearing regions of the jaws. The areas affected by secondary osteomyelitis show radiolucent zones surrounding the radiopaque lesions (Figure 1). The first patient presented with a jaw fracture following a failed extraction attempt (Figure 1- arrow). Due to the extensive bony sclerosis, associated impacted teeth are common (Figure 2).
Genetic conditions including hyperparathyroidism-jaw tumour syndrome (HPT-JT), gnathodiaphyseal dysplasia (GDD) and familial florid cemento-osseous dysplasia (FFCOD) must be considered as part of the differential diagnoses. Following strict clinicopathological correlation, as well as the patients lacking a history of co-morbidities and long bone fractures, both HPT-JT and GDD were excluded.
FFCOD has a similar radiographic presentation to conventional florid cemento-osseous dysplasia with subtle differences. They are similar in that they present with mixed radiolucent-radiopaque lesions limited to the tooth-bearing regions of the jaws, characteristically affecting multiple quadrants. The conditions differ in that the familial variant presents at a younger age with an earlier onset of bone sclerosis, an increased propensity for expansion, and multiple tooth impactions. Additionally, the familial variant shows a decreased predilection for females. Both variants have a risk for the development of secondary osteomyelitis due to the hypovascularity of the bone. Therefore, optimal oral hygiene and preventive measures ensuring tooth retention must be prioritised. Extractions should be avoided as far as possible to reduce the risk of osteomyelitis, which is extremely difficult to treat in these instances.1
These cases highlight the importance of radiographic imaging in the diagnosis of benign fibro-osseous lesions and for thorough case workup before dental interventions are performed.
REFERENCES
1. Nel C, Yakoob Z, Schouwstra CM, van Heerden WF. Familial florid cemento-osseous dysplasia: a report of three cases and review of the literature. Dentomaxillofacial Radiology. 2021 Jan 1;50(1):20190486. [ Links ]
2. Sim YC, Bakhshalian N, Lee J-H, Ahn K-M. Familial florid cemento-osseous dysplasia in mother and her identical twins : a report with review of the literatures. Oral Surg. 2014;7:239-44. [ Links ]
Correspondence:
Chané Smit
Department of Oral Pathology and Oral Biology
University of Pretoria. Tel +27 (0)12 319 2311
Email: chane.smit@up.ac.za
Authors contribution:
Chané Smit: 70%
Zarah Yakoob: 30%