Open Access
Issue
Med Buccale Chir Buccale
Volume 23, Number 4, December 2017
Page(s) 196 - 199
Section Cas clinique et revue de la littérature / Up-to date review and case report
DOI https://doi.org/10.1051/mbcb/2017017
Published online 22 December 2017

© The authors, 2017

Licence Creative CommonsThis is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Introduction

Bone dysplasia, formerly known as cemento-osseous dysplasia, is characterized by benign bony idiopathic lesions. It originates from the alveolar ligament and is characterized by the replacement of the normal bone by fibrous tissue and metaplastic bone tissue [1,2]. It is divided into four forms depending on the location, size, and number of lesions: (i) periapical bone dysplasia involves a limited number of teeth and is located in the anterior part of the mandible or maxilla; (ii) focal bone dysplasia also involves a limited number of teeth but has a posterior localization; (iii) florid bone dysplasia involves several quadrants; (iv) the gigantiform cementoma is a single lesion that can reach a large size and primarily affects children [3,4]. These lesions are usually asymptomatic and are often discovered incidentally on panoramic X-rays. Symptom onset coincides with an increase in size or intraoral exposure of dysplastic lesions during infectious episodes. Here we discuss the case of a patient with a florid osseous dysplasia associated with neurosensory disturbance of the inferior alveolar nerve in the labiomental region.

Observation

A 45-year   istory. Her calcium/phosphate equilibrium was normal. The intraoral examination found firm and painless vestibular swelling of the mandibule with a normal mucosa (Fig. 1), and the vitality pulp testing was positive for all teeth near the bone dysplasia. The panoramic X-ray showed apical lesions of the teeth 47, 45, 44, 43, 42, 41, 31, 32, 33, 34, 35, 37 as well as decay or restorative treatments of the teeth 48, 47, 44, 37, and 38 (Fig. 2). Cone-beam computerized tomography (CBCT) has allowed a more precise characterization of the appearance, size, and anatomical ratios of the various lesions around the teeth as well as those in close contact with the mental foramen and the right mandibular foramen (Fig. 3a and b). These lesions were bilateral and symmetrical, multilobulated, irregular in shape, of mixed appearance, and had clear margins. Florid osseous dysplasia was diagnosed on the basis of these scans. Conservative monitoring was established on an annual basis. Six month later, the patient consulted after the occurence of paresthesia as burning and tingling sensation and pain in the right labiomental region. Yet, any infection of the lesions were observed. Following the altered symptomatology, a root canal treatment of the teeth [5] in contact with the lesions was performed under antibiotic prophylaxis then a resection of the sequestered cementoosseous was done under general anesthesia (Fig. 4). The panoramic X-ray performed following the surgery confirmed the complete resection of the lesions (Fig. 5). The pathological examination confirmed the diagnosis of cementomas with the presence of irregular pagetoid osteoids of different shapes, sizes, and thicknesses with irregular cement lines. Osteoblasts were present in contact with the newly formed bone. Bone segments were filled with benign fibroblast proliferations. One month after surgery, the patient showed good wound healing, the neuropathic pain had disappeared but there was still an isolated numbness of the oral mucosa compared to the controlateral area. Six months after surgery, the hypoesthesia had almost disappeared; the panoramic X-ray showed a gradual filling of cavities and a lack of recurrence (Fig. 6).

thumbnail Fig. 1

Left mandibular buccal bone swelling.

thumbnail Fig. 2

Initial panoramic radiography with mixed multilobular well-defined images of the alveolar bone, respecting the basilar bone.

thumbnail Fig. 3

CBCT initial scan: irregular radiopacity lesions surround the apical region of tooth 47 (black arrow) and near by the inferior alveolar nerve foramen (white arrow). (a) Coronal cut. (b) Sagittal cut near by the tooth 47.

thumbnail Fig. 4

Bone cavities after osseous dysplasia resection.

thumbnail Fig. 5

Post-operative panoramic radiography.

thumbnail Fig. 6

Six months post-op panoramic radiography: a gradual mineralization of the intra-osseous cavity is obvious.

Comments

Florid osseous dysplasia is a rare and benign condition characterized by a multifocal onset that can affect all four quadrants of the mouth. It mainly affects women of African origin between the ages of 40 and 50 years [6,7]. Although the etiology and the triggering factors were not identified [8], Florid osseous dysplasia could result from abnormal activity of the periodontal ligament explaining its near-exclusive location in the dental region, with cemento-osseous lesions identified in relation to dental apices and dental anatomy in radiographic examinations [9]. It is generally asymptomatic and the teeth in relation to the lesions are unaffected [1012]. Florid osseous dysplasia becomes symptomatic when lesions are accompanied by infection, or if cortical fenestrations occur [3]. The diagnosis is radiological with the presence of mixed multilobular well-defined images of the alveolar bone, respecting the basilar bone.Any rhizalysis are reported [11]. Other diagnoses are ossifying fibroma, Paget disease, diffuse chronic osteomyelitis, Gardner syndrome, and endodontic lesions [12]. In the absence of symptomatology, it is recommended to conduct annual clinical and radiological monitoring and to maintain rigorous oral hygiene to avoid infections [13]. However, because of their sclerotic and vascular nature, lesions are particularly susceptible to infection after mucosal trauma, dental procedures, biopsy or cystic enucleation [14,15]. This risk of iatrogenic infection can justify an antibiotic prophylaxis in any surgical procedure [16]. The infection is accompanied by  soft-tissue swelling and pain but without any hypoesthesia in the territory of the inferior alveolar nerve [13,15,1719].

In the presented case, the pain has occured without any infectious process or mucosal lesion. The symptoms are typical of neuropathic pain with burning sensation, tingling and electric shocks of the lower lip. According to the Kawai classification, CBCT unveiled stage-E lesions (irregular radio-opaque lesions surrounding the dental root) and stage-D lesions (lobular or spherical calcified masses surrounded by a radiolucent border) around the roots of the teeth 46, 47, and 48, which is typical of florid osseous dysplasia [20]. The proximity between the calcified masses and the mental canal or mandibular foramen could justify the sensitive disorders.

In the absence of bony sequestrum, surgery is difficult because the lesions can not be easily separated from the healthy bone and its is necessary to perform curettage of the alveolar bone reach healthy bone [21]. Some authors recommend that cortical fenestrations be performed in lesions to induce better healing of bone tissue [22]. When faced with the cleavable aspect of the apical cemento-osseous lesions and the need for a diagnosis, we opted for a surgical resection of the lesions and a grinding of the cavity walls after enucleation.

Despite a lack of consensus regarding this, in cases of multifocal lesions in the presence of decayed or treated teeth or the risk of pulp necrosis or infection after a surgery, endodontic treatment of all the teeth in contact with the lesions is recommended. The two procedures (endodontic treatment and lesion resection) involved an antibiotic prophylaxis because the literature reported a case of osteomyelitis after an endodontic treatment in a patient with florid osseous dysplasia [23].

Conclusion

There is no consensus on the management of florid osseous dysplasia. Nevertheless, some guidelines are found in the literature. If the patient is asymptomatic, clinical and radiological follow-up must be instituted. If the patient has an infection, an antibiotic treatment and sometimes a resection of sequestered cemento-osseous apices should be proposed. In case of neuropathic pain, the anatomical decompression of the lower alveolar nerve relieves the pain.

Conflicts of interests

The authors declare that they have no conflicts of interest in relation to this article.

Acknowledgments

J.-D. Souyris for the endodontic treatments performed and to Romy Makhoul for the English corrections.

References

  1. White SC, Pharoah MJ. Oral radiology: principles and interpretation, 5th edn., St Louis: Mosby, 2004:485–498. [Google Scholar]
  2. Coleman H, Altini M, Kieser J, Nissenbaum M. Familial florid cemento-osseous dysplasia a case report and review of the literature. J Dent Assoc South Afr 1996;51:766–770. [Google Scholar]
  3. Thompson L. World Health Organization classification of tumours: pathology and genetics of head and neck tumours. Ear Nose Throat J 2006;85:74. [PubMed] [Google Scholar]
  4. Barnes L, Eveson JW, Reichart P, Sidransky D. World Health Organization classification of tumours. Pathology and genetics of head and neck tumours, Lyon: IARC Press, 2005. [Google Scholar]
  5. Kim S, Kratchman S. Modern endodontic surgery concepts and practice: a review. J Endod 2006;32:601–623. [CrossRef] [PubMed] [Google Scholar]
  6. MacDonald-Jankowski DS. Florid cemento-osseous dysplasia: a systematic review. Dento Maxillo Facial Radiol 2003;32:141–149. [CrossRef] [PubMed] [Google Scholar]
  7. Melrose RJ, Abrams AM, Mills BG. Florid osseous dysplasia. A clinical-pathologic study of thirty-four cases. Oral Surg Oral Med Oral Pathol 1976;41:62–82. [CrossRef] [PubMed] [Google Scholar]
  8. Wright JM. Reactive, dysplastic and neoplastic conditions of periodontal ligament origin. Periodontology 1999;21:7–15. [CrossRef] [PubMed] [Google Scholar]
  9. Khonsari RH, Corre P, Bougila J, Gayet-Delacroix M, Piot B. Lésions péri-apicales multiples en cible. Rev Stomatol Chir Maxillofac 2008;109:403–404. [CrossRef] [PubMed] [Google Scholar]
  10. Delai D, Bernardi A, Felippe GS, da Silveira Teixeira C, Felippe WT, Santos Felippe MC. Florid cemento-osseous dysplasia: a case of misdiagnosis. J Endod 2015;41:1923–1926. [CrossRef] [Google Scholar]
  11. Alsufyani NA, Lam EWN. Osseous (cemento-osseous) dysplasia of the jaws: clinical and radiographic analysis. J Can Dent Assoc 2011;77:b70. [PubMed] [Google Scholar]
  12. Massereau E, Ordioni U, Guivarc'h M, Royer G, Catherine J-H. Dysplasie osseuse floride mandibulaire: un cas de découverte fortuite et revue de la littérature. Méd Buc Chir Buc 2015;21:101–104. [CrossRef] [Google Scholar]
  13. Gerlach RC, Dixon DR, Goksel T, Castle JT, Henry WA. Case presentation of florid cemento-osseous dysplasia with concomitant cemento-ossifying fibroma discovered during implant explantation. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;115:e44–e52. [CrossRef] [Google Scholar]
  14. Sadda RS, Phelan J. Dental management of florid cemento-osseous dysplosia. NY State Dent J 2014;80:24–26. [Google Scholar]
  15. Fenerty S, Shaw W, Verma R, Syed AB, Kuklani R, Yang J et al. Florid cemento-osseous dysplasia: review of an uncommon fibro-osseous lesion of the jaw with important clinical implications. Skeletal Radiol 2017;46:581–590. [CrossRef] [PubMed] [Google Scholar]
  16. Wolf J, Hietanen J, Sane J. Florid cemento-osseous dysplasia (gigantiform cementoma) in a caucasian woman. Brit J Oral Maxil Surg 1989;27:46–52. [CrossRef] [Google Scholar]
  17. Heuberger BM, Bornstein MM, Reichart PA, Hürlimann S, Kuttenberger JJ. Periapical osseous dysplasia of the anterior maxilla – a case presentation. Schweiz Monatsschr Zahnmed 2010;120:1001–1011. [PubMed] [Google Scholar]
  18. Maccotta M, Radoï L. Florid osseous dysplasia: management of a symptomatic case. Rev Stomatol Chir Maxillo-Faciale Chir Orale 2016;117:425–428. [CrossRef] [Google Scholar]
  19. Lahbabi M, Fleuridas G, Lockhart R, Delabrouhe C, Guilbert F, Bertrand JC. Florid cemento-osseous dysplasia: a poorly recognized entity. Report of 5 cases. Rev Stomatol Chir Maxillofac 1998;99:33–39. [PubMed] [Google Scholar]
  20. Kawai T, Hiranuma H, Kishino M, Jikko A, Saduka M. Cemento-osseous dysplasia of the jaws in 54 Japanese patients. A radiographic study. Oral Surg Oral Med Oral Patho Oral Radio Endod 1999;87:107–114. [CrossRef] [Google Scholar]
  21. Moussaoui E, Oualha L, Gnaba I, Ayachi S, Sriha B, Selmi J, Douki N. Dysplasies osseuses florides : du diagnostic au traitement. A propos de trois cas. Med Buccale Chir Buccale 2013;19:119–126. [CrossRef] [EDP Sciences] [Google Scholar]
  22. Fauroux MA, Menjot de Champfleur S, Torres JH. Simple bone cyst associated with florid osseous dysplasia: 2 case reports. Med Buc Chir Buc 2014;20:41–45. [CrossRef] [Google Scholar]
  23. Schneider LC, Mesa ML, Brickman JH. Complications of endodontic therapy in florid osseous dysplasia. Oral Surg Oral Med Oral Pathol 1987;64:114–116. [CrossRef] [PubMed] [Google Scholar]

All Figures

thumbnail Fig. 1

Left mandibular buccal bone swelling.

In the text
thumbnail Fig. 2

Initial panoramic radiography with mixed multilobular well-defined images of the alveolar bone, respecting the basilar bone.

In the text
thumbnail Fig. 3

CBCT initial scan: irregular radiopacity lesions surround the apical region of tooth 47 (black arrow) and near by the inferior alveolar nerve foramen (white arrow). (a) Coronal cut. (b) Sagittal cut near by the tooth 47.

In the text
thumbnail Fig. 4

Bone cavities after osseous dysplasia resection.

In the text
thumbnail Fig. 5

Post-operative panoramic radiography.

In the text
thumbnail Fig. 6

Six months post-op panoramic radiography: a gradual mineralization of the intra-osseous cavity is obvious.

In the text

Current usage metrics show cumulative count of Article Views (full-text article views including HTML views, PDF and ePub downloads, according to the available data) and Abstracts Views on Vision4Press platform.

Data correspond to usage on the plateform after 2015. The current usage metrics is available 48-96 hours after online publication and is updated daily on week days.

Initial download of the metrics may take a while.