Research

Atypical Presentation of Florid Osseous Dysplasia with Extensive Multifocal Expansion and Simple Bone Cyst

Joshua Orgill DDS
Resident, Oral and Maxillofacial Radiology
Department of Oral Pathology, Radiology and Medicine 1,2
The College of Dentistry and Clinics,
The University of Iowa
joshua-orgill@uiowa.edu
Ph: 319-335-7362

Address for correspondence

Sindhura Anamali B.D.S., M.S
Diplomate of the American Board of Oral and Maxillofacial Radiology
Clinical Assistant Professor
Department of Oral Pathology, Radiology and Medicine 1,2
The College of Dentistry and Clinics,
The University of Iowa
sindhura-anamali@uiowa.edu
Ph: 319-335-8956

Published on : 24 Jan 2019

Abstract:

Florid osseous dysplasia (FOD), is a broad form of periapical osseous dysplasia (POD) involving multiple quadrants {Neville, 2015 #36} . Like POD, FOD occurs predominantly in middle-aged females with a significant predilection for African Americans and to a lesser extent in Asians. This condition is characterized by replacement of normal cancellous bone with fibrous tissue and dense amorphous bone. This case presents the findings of a CBCT that was obtained on a 58 year old African American female and reviewed by a board- certified oral and maxillofacial radiologist. The findings were of an atypical presentation of florid osseous dysplasia with multifocal expansion and simple bone cysts. Though florid osseous dysplasia is not an uncommon finding, the extent and presentation of this case is unusual and has definite educational value as it shows the extent of progression of a more severe case and the important role of clinicians in the diagnosis and treatment planning.

Keywords :

Introduction:

With the introduction and acceptance of CBCT use in dentistry the ability to more completely visualize the jaws has increased. With this increased ability to visualize the anatomical structures of the jaws there is also an increased ability to visualize pathological conditions. Greater radiographic insight of various pathoses have led to greater diagnostic abilities. 1 Visualization of relationships between pathoses and anatomic structures has also been a great benefit by the use of CBCT giving dentists and surgeons more certainty to treatment options and better treatment planning. severe case and the important role of clinicians in the diagnosis and treatment planning.

Osseous dysplasia (OD) is a non-neoplastic condition where normal bone is replaced with fibrous tissue and amorphous bone or abnormal bone trabeculae. 2 OD is considered to be the most common fibro-osseous lesion of the jaws. 3 As described in the 2005 publication of World Health Organization (WHO), OD has 3 clinical presentations which include; periapical osseous dysplasia, focal osseous dysplasia, and florid osseous dysplasia. 4 Alsufyani and Lam described in a study that looked at 118 patients with OD that 78.8% were of the periapical osseous dysplasia type while the other 21.2% were of the florid osseous dysplasia type. 1 Demographics for OD is reported as being most common in black females, from 82.9% to over 90%, and in their fifth decade of life. 1-4

3 stages of OD have been described to include the osteolytic stage; a radiolucent appearing stage, the cementoblastic stage; a radiolucent area with radiopaque zones, and the mature stage; with definite radiopaque masses in the majority of the lesion. 5 Careful radiographic diagnosis is important, because biopsy and surgery are typically undesirable and might lead to complications including poor healing and osteomyelitis. 6, 7

Case report:

The case reported is of a 58 year old African American female who presented with an enlargement of the mandible. She did not report any family history of such a presentation, her health history was unremarkable. A CBCT was made on an i-CAT Next generation machine (Imaging Sciences International, Hartsfield, PA), the field of view was 16cm X 13cm made at 0.25mm resolution and was reviewed by a board-certified oral and maxillofacial radiologist. Among the findings noted were multiple expansile, mixed radiolucent/radiopaque entities present bilaterally in both the maxillae and the mandible. There was extensive involvement of the mandible from the mandibular right third molar to the mandibular left third molar regions with displacement of the inferior alveolar canals inferiorly, as well as extensive expansion of both the facial and lingual cortical plates. The cortices of the mandible were maintained albeit with very thin borders. The mandibular anterior teeth have been minimally displaced with no evidence of resorption of the roots. In the posterior aspect of both the right and left maxillae there was expansion of the cortices in the facial and palatal directions. The cortices were maintained with minimal evidence of tooth displacement and no evidence of root resorption. There was displacement of the floor of the right maxillary sinus. There were simple bone cysts in the apical regions of the maxillary right first molar and bilaterally in the right and left body and anterior region of the mandible. Incidental findings included generalized minimal periodontal bone loss, a mucus retention pseudocyst on the lateral wall of the left maxillary sinus, mild degenerative changes of the cervical vertebrae, and multiple carotid artery calcifications bilaterally in both the parasellar region and in the neck region. Considering the nature of the condition it was not warranted to biopsy at the time of the examination. Patient has been advised and educated to adapt good oral hygiene practices, prophylaxis and regular follow up to prevent possible complications with this condition. A follow up was scheduled in a year unless in the unlikely scenario the patient became symptomatic.

Discussion:

Osseous dysplasias are relatively a frequent condition occurring in the jaws in black females typically in the 4 th to 6 th decades of life. 6-10 Typical cases of osseous dysplasia have been frequently and comprehensively described in the literature. They are generally categorized into periapical, focal and florid types 11 , based on the presentation and scope of findings visualized, however there are very few cases which have been reported which exhibit such extreme findings as in this case. The prevalence of these variants are periapical (57.3%), focal (28%), and florid (14.6%) and other extreme variants less than 0.1% of the cases. 11 Periapical osseous dysplasias are in the apical regions of anterior teeth, focal variants are usually associated with a single tooth and the florid variants are those in which there is multifocal involvement with lesions in more than one quadrant in the jaws. These entities may be radiolucent in the early stages, mixed radiolucent radiopaque in the intermediate stage and almost completely radiopaque in the mature stages. 8, 9, 12

Florid cemento osseous dysplasias (FCOD) have been reported in the past, only few cases of severe expansile osseous dysplasias have been presented in the literature. FCOD have a predilection for African American and middle age females. The characteristics of FCOD in the initial stages are similar to those of periapical lesions of inflammatory origin, which may lead to misdiagnosis. 13

Steven R Singer et al 14 have presented a similar expansile case of florid expansile cemento- osseous dysplasia but without simple bone cysts. A similar expansile presentation can be seen in cases of Gigantiform cementoma, but in our case the patient did not have a family history of such a presentation and if biopsied histopathologically Florid osseous dysplasia and Gigantiform cementoma would present similar features and it would be difficult to separate one entity from the other (Florid osseous dysplasia from Gigantiform) cementoma 3 . The surgical management of Gigantiform cementoma is usually difficult because of the extensive involvement of the these tumors in multiple quadrants and the location of these tumors usually pose an inadequate access to completely remove them 15 . The surgical treatment for Gigantiform cementomas with extensive involvement is not successful and tends to have a high recurrence rate 15 .

Florid osseous dysplasias can present in a wide spectrum from initial radiolucent stages to intermediate mixed radiopaque –radiolucent lesions to completely mature radiopaque stage in multiple quadrants. Some lesions can cause significant expansion and can cause facial disfigurement and may be accompanied with simple bone cysts. Early detection and correct diagnosis is prudent in these patients to prevent possible complications that could result from unnecessary work up such as biopsies, improper diagnosis and treatment planning.

Due to the decreased vascularity within areas of FOD it is important to make a proper radiographic diagnosis thus preventing intervention which may unnecessarily increase the risk of infection. 2, 3, 5 It is important for clinicians to be well informed that a biopsy is contraindicated in these lesions as it can make the condition worse. Conservative approaches such as broad spectrum antibiotics in case of infections along with periodic follow-up and good oral care is advised. 16 Caution in identification and diagnosis should be taken, as unnecessary treatment, such as root canal therapy, may result when it should be avoided. 17-19 Good oral hygiene practices should be advised to the patient to prevent secondary infection. 2, 3, 5, 20, 21 No treatment is necessary unless it becomes a cosmetic concern.

Conflict of Interest: None

References:

  1. Alsufyani NA, Lam E. Osseous (cemento-osseous) dysplasia of the jaws: clinical and radiographic analysis. J Can Dent Assoc. 2011;77:b70.
  2. White SC, Pharoah MJ. Oral Radiology-E-Book: Principles and Interpretation: Elsevier Health Sciences; 2014.
  3. Neville BW, Damm DD, Chi AC, Allen CM. Oral and maxillofacial pathology: Elsevier Health Sciences; 2015.
  4. Barnes L. Pathology and genetics of head and neck tumours: IARC; 2005.
  5. Yildirim E, Bağlar S, Ciftci ME, Ozcan E. Florid cemento-osseous dysplasia: A rare case report evaluated with cone-beam computed tomography. Journal of oral and maxillofacial pathology: JOMFP. 2016;20:329.
  6. MacDonald-Jankowski DS. Florid cemento-osseous dysplasia: a systematic review. Dentomaxillofac Radiol. 2003;32:141-149.
  7. Macdonald-Jankowski DS. Focal cemento-osseous dysplasia: a systematic review. Dentomaxillofac Radiol. 2008;37:350-360.
  8. Alsufyani NA, Lam EW. Osseous (cemento-osseous) dysplasia of the jaws: clinical and radiographic analysis. J Can Dent Assoc. 2011;77:b70.
  9. Alsufyani NA, Lam EW. Cemento-osseous dysplasia of the jaw bones: key radiographic features. Dentomaxillofac Radiol. 2011;40:141-146.
  10. Chadwick JW, Alsufyani NA, Lam EW. Clinical and radiographic features of solitary and cemento-osseous dysplasia-associated simple bone cysts. Dentomaxillofac Radiol. 2011;40:230-235.
  11. Gale N, Poljak M, Zidar N. Update from the 4th Edition of the World Health Organization Classification of Head and Neck Tumours: What is New in the 2017 WHO Blue Book for Tumours of the Hypopharynx, Larynx, Trachea and Parapharyngeal Space. Head Neck Pathol. 2017;11:23-32.
  12. Waldron CA. Fibro-osseous lesions of the jaws. J Oral Maxillofac Surg. 1993;51:828- 835.
  13. 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.
  14. Singer S, Creanga A, Vyas R, Mupparapu M. Florid expansile cemento-osseous dysplasia of the jaws: Cone beam computed tomography study and review of the literature. Journal of Orofacial Sciences. 2017;9:114-117.
  15. Abdelsayed RA, Eversole LR, Singh BS, Scarbrough FE. Gigantiform cementoma: Clinicopathologic presentation of 3 cases. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 2001;91:438-444.
  16. Thorawat A, Kalkur C, Naikmasur VG, Tarakji B. Familial florid Cemento-osseous dysplasia - case report and review of literature. Clin Case Rep. 2015;3:1034-1037.
  17. Delai D, Bernardi A, Felippe GS, da Silveira Teixeira C, Felippe WT, Felippe MCS. Florid Cemento-osseous Dysplasia: A Case of Misdiagnosis. Journal of endodontics. 2015;41:1923-1926.
  18. Huh J-K, Shin S-J. Misdiagnosis of florid cemento-osseous dysplasia leading to unnecessary root canal treatment: a case report. Restorative dentistry & endodontics. 2013;38:160-166.
  19. Rekabi AR, Ashouri R, Torabi M, Parirokh M, Abbott PV. Florid cemento‐osseous dysplasia mimicking apical periodontitis: A case report. Australian Endodontic Journal. 2013;39:176-179.
  20. Fenerty S, Shaw W, Verma R, et al. Florid cemento-osseous dysplasia: review of an uncommon fibro-osseous lesion of the jaw with important clinical implications. Skeletal radiology. 2017;46:581-590.
  21. Mainville GN, Turgeon DP, Kauzman A. Diagnosis and management of benign fibro‐osseous lesions of the jaws: a current review for the dental clinician. Oral diseases. 2016.

Image 1

Multi-planar reconstruction images; showing expansile multiple mixed radiopaque-radiolucent areas in the mandible with simple bone cysts.

Image 2

Multi-planar reconstruction images; showing expansile multiple mixed radiopaque-radiolucent areas in the mandible and maxilla with simple bone cysts.

Image 3

Coronal slice CBCT image; showing expansile mixed radiopaque radiolucent areas in the right maxilla and in the anterior mandible.

Image 4

Coronal slice CBCT image; showing expansile mixed radiopaque radiolucent areas in all the four quadrants; maxilla and the mandible.

Image 5

Sagittal slice CBCT image; showing expansile mixed radiopaque-radiolucent area with simple bone cysts in the anterior mandible.

Image 6

Axial slice CBCT image; showing multiple expansile mixed radiopaque- radiolucent areas with simple bone cysts in the mandible with extensive involvement.

Image 7

Volume rendered CBCT image; showing multiple expansile and lobulated radiopaque areas in the right side of the mandible and the maxilla.

Image 8

Volume rendered CBCT image, showing multiple expansile and lobulated radiopaque areas in the left side of the mandible.

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