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CASE REPORT
Year : 2018  |  Volume : 3  |  Issue : 1  |  Page : 33-38

Cemento-osseous dysplasia in the anterior mandible: A case of misdiagnosis


Restorative Department Science, College of Dentistry, King Khalid University, Abha, Kingdom of Saudi Arabia

Date of Web Publication13-Aug-2020

Correspondence Address:
BDS, MS Abdulaziz S Abu-Melha
Restorative Department Science, College of Dentistry, King Khalid University
Kingdom of Saudi Arabia
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Source of Support: None, Conflict of Interest: None


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  Abstract 


Osseous dysplasia (Cemento-osseous dysplasias) is relatively common form of fibro osseous lesions (FOLs) in the tooth-bearing regions of the jaw bones. Clinically, most of the Osseous dysplasias (ODs) are asymptomatic and are accidently discovered during routine radiographic examinations. A proper diagnosis of OD is very crucial to avoid misdiagnosis and mismanagement. The histological appearance is similar to other fibro osseous lesions (FOLs) and in numerous cases; Osseous dysplasias have been misdiagnosed as periapical lesions (Periapical abscess, granuloma or cyst), and/or mismanaged by initiation of unnecessary endodontic and surgical interventions. We report a misdiagnosed case of Focal Cemento-Osseous Dysplasia and unnecessary endodontic treatment in a 48 years old woman due to similar radiographic appearance of the lesions during the osteolytic phase of Osseous Dysplasia. This unnecessary invasive treatment could have been minimized or avoided if standard protocols of pulp vitality and a radiographic interpretation were performed carefully.

Keywords: Focal Cemento -osseous dysplasia, Misdiagnosis, Periapical pathology, Pulp vitality


How to cite this article:
Abu-Melha AS. Cemento-osseous dysplasia in the anterior mandible: A case of misdiagnosis. King Khalid Univ J Health Scii 2018;3:33-8

How to cite this URL:
Abu-Melha AS. Cemento-osseous dysplasia in the anterior mandible: A case of misdiagnosis. King Khalid Univ J Health Scii [serial online] 2018 [cited 2020 Dec 3];3:33-8. Available from: https://www.kkujhs.org/text.asp?2018/3/1/33/291946




  Introduction Top


The term fibro osseous lesions (FOLs) refers to a group of poorly defined lesions affecting the jaws and cranio facial bones, characterised by the replacement of normal bone with fibrous connective tissue matrix composed of collagen fibers and fibroblasts and containing varying amounts of mineralized substances such as osteoid, psammoma body, mature bone or cementum like calcifications.[1] Although the histopathology of these fibro-osseous lesions (FOLs) are similar in appearance, they have a very different clinical, radiological presentations, behaviour, and treatment outcomes.

Osseous dysplasia (Cemento-osseous dysplasias) is the most common form of FOL in the tooth-bearing regions of the jaw bones.[2] They are considered as benign odontogenic lesions of the jaws with abnormal bone as its composition and not a form of cementum.[3] It is hypothesized to originate by the proliferation of cells in the periodontal ligament &/or periodontium. Historically these lesions were referred to as cemento-osseous dysplasias and believed to be reactive in origin. The understanding of fibro-osseous lesions has changed over the last decade and is now referred to as osseous dysplasias with”cemento-osseous dysplasia” (COD) as a synonym.

Based on its clinical and radiographic features, OD/COD is mainly divided into 3 subtypes: periapical cemento-osseous dysplasia (POD), focal cement-osseous dysplasia (Focal OD), and florid cemento-osseous dysplasia. For a more simplistic understanding, the previous WHO guidelines were modified by Su et al.(1997) proposing use of the term Focal OD for lesions occurring in the anterior mandible, other parts of the jaws and Florid OD for similar lesions involving more than one quadrant in the jaws.[4],[5],[6]

Clinically, most of the OD are asymptomatic and are routinely discovered during routine radiographic examinations. Rarely, large lesions do occur and can be associated with bony expansion, pain and facial deformity. A proper diagnosis of OD is very crucial to avoid misdiagnosis and mismanagement. The histological appearance is similar to other FOLs and in numerous cases; OD have been misdiagnosed as periapical lesions (Periapical abscess, granuloma or cyst), and/or mismanaged by initiation of unnecessary endodontic treatment, due to failure of the specialist to identify these lesions in their early stages that appear radiolucent at the apical region of the root. The mature lesions have been misdiagnosed as chronic sclerosing osteomyelitis, cement-ossifying fibroma, odontoma and osteoblastoma leading to unnecessary surgical interventions.[7],[8],[9],[10] Radiographically, these lesions commonly manifest in the apical areas of the teeth as radiolucent mass in the initial osteolytic phase, followed by a blastic phase when lesions appear mixed radiolucent and radiopaque to osteogenic radiopaque mass surrounded by a radiolucent peripheral ring in the final stage of the lesion.[11]

We report a case of unnecessary root canal treatment of teeth presenting with periapical OD/COD due to a misdiagnosis as periapical pathology, the subsequent management of the case highlighting the need for correct diagnosis and avoidable endodontic treatment.


  Case Report Top


A 48 year middle aged woman visited to the department of Restorative Dental Science, King Khalid University, Abha; with a chief complaint of pain and discomfort since over a month in the mandibular front tooth region. The patient had a significant dental history of pain and discomfort in the mandibular left central incisor, two months ago and was treated by root canal treatment for the same by her general dentist. The symptoms weren’t relieved despite the root canal treatment and the patient was referred to the endodontist for further evaluation. The patient was otherwise healthy with no significant medical history and all her general vital signs were within normal limits. Extra oral findings were within normal limits and were negative for any submandibular or cervical lymphadenopathy. Examination of the oral cavity demonstrated poor hygiene and several restorations. The labial vestibule in relation to the mandibular left central incisor was tender on palpation and was negative for any swelling or sinus tract. Periodontal examination showed probing depths within normal limits.

The Mandibular left lateral incisor showed delayed response to electric pulp testing in comparison to normal response of mandibular right central and lateral incisors. An IOPA radiograph revealed an adequately root canal filled 31 with a well defined mixed radiolucent radiopaque mass in the apical region of the mandibular left central and lateral incisors with normally appearing periodontal ligament space and continuous lamina dura in the apical third of the incisors [Figure 1].
Figure 1: Radiograph showing adequately filled Root canal of mandibular left central incisor with periapical radioluceny

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A provisional diagnosis of Non Pulpal pathology was made. Re-RCT of mandibular left central incisor was performed [Figure 2] and the patient was administered analgesics and follow up review after six months, unless worsening symptoms required immediate intervention. A follow up review after six months revealed lack of pain or tenderness on palpation and the mandibular left lateral incisor showed delayed response to electric heat vitality test. An occlusal access without anesthesia was performed in mandibular left lateral incisor and was found to be vital. The occlusal cavity was duly restored and required no further intervention [Figure 2].
Figure 2: Radiograph showing Re treated mandibular left central incisor and occlusal restoration of mandibular left lateral incisor

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A follow –up radiograph taken after 6 months demonstrated minor reduction in the size of the well defined mixed radiolucent and radiopaque lesion with more radio density than the previous one [Figure 3].
Figure 3: Follow –up radiograph taken after 6 months showed a minor reduction in the size of the well defined mixed radiolucent and radiopaque lesion with more radio density.

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A CBCT study was done that revealed mixed density lesions in relation to mandibular left central and lateral incisors and mandibular left third molar with mild cortical resorption and expansion in the affected areas, suggestive of Fibro osseous lesion [Figure 4].
Figure 4: CBCT images showing mixed density lesions in relation to mandibular left central and lateral incisors and mandibular left third molar with mild cortical resorption and expansion in the affected areas.

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An exploratory biopsy was performed from the perforated bone and a mix of bone and tissue was sent for histopathological examination which revealed features of cellular fibrous connective tissue mixed with osteoid trabaculae consisting of osteoblastic rimming and small irregular and round calcified areas suggestive of Benign Fibro osseous lesion [Figure 5]a, [Figure 5]b. The patient was informed of the same and was advised for periodic follow-up.
Figure 5a & b: Histopathological sections showing features of cellular fibrous connective tissue mixed with osteoid trabaculae consisting of osteoblastic rimming and small irregular and round calcified areas

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  Discussion Top


Osseous dysplasia (OD/CODs) is the most common form of FOL in the tooth-bearing regions of the jaw bones.[2] The exact etiopathogenesis of Focal OD is unknown but is believed to originate from the periodontal ligament space, the medullar bone or both. They are classified as benign odontogenic lesions of the jaws with abnormal bone as its primary composition and not a form of cementum (as earlier believed).[3]

Based on their clinico-radiographic presentations; their specific location and type of distribution (isolated or multiple lesions), three main sub-types of ODs namely focal, periapical, florid and a specific rare variant, the familial gigantiform cementoma have been described. They have similar origin and pathological process with distinct clinic- radiographic appearance. The term “Focal OD is used for lesions involving the mandibular anterior teeth and other areas as solitary lesions” [5],[6] Radiographically, these lesions initially appear as osteolytic radiolucent areas that mature over time and become progressively radiopaque described under 3 radiographic stages as below: Stage 1 - osteolytic phase seen as radiolucent mass, Stage 2 - mixed radiolucent radiopaque mass, Stage 3 - osteogenic phase seen as radiopaque mass.

ODs/CODs are usually asymptomatic and discovered during routine radiographic examination. The diagnosis of ODs are challenging and confusing due to overlapping features associated with FOLs. The distinct clinical and radiologic features help in differential diagnosis and a most plausible diagnosis can be made by noting the stage at which these lesions appear on radiographic examination and by ruling out the commonly similar lesions such as osteomyelitis of the jaws and traumatic bone cysts. However, several cases of misdiagnosis and mismanagement of these lesions have been reported widely in literature.

ODs can occur at any age, but it is reportedly more common among middle-aged women of Asian, East Asian or African descent and more common in women. Most of the asymptomatic cases of OD tend to occur among younger individuals and discovered during routine radiographic examination in contrast to older individuals presenting with one or more clinically evident symptoms similar to our case report.[5],[13],[14],[15],[16]

Clinical manifestations include pain, swollen gingival, and purulent discharge, tenderness on palpation or delayed wound healing after extraction and secondary infection mimicking osteomyelitis in the absence of an obvious dental cause.[17]

In this report, we have described a case Focal OD that was initially misdiagnosed as inflammatory periapical lesion and an unnecessary endodontic treatment was performed in a middle aged woman due to similar radiographic appearance of the lesions during the osteolytic phase of OD. This unnecessary invasive treatment could have been minimized/avoided if standard protocols of pulp vitality and a radiographic interpretation were performed carefully.

OD/CODs have typical radiographic appearance and can be differentiated from similar periapical lesions on careful clinical examination and radiological correlation. Focal OD occur frequently in mandibular teeth as well defined radiolucent lesions in the initial stages followed by mixed radiolucent and radiopaque structure and completely radiopaque with a peripheral radiolucent rim at mature stages as seen in our case during follow up radiographic examination. Establishing pulp vitality is extremely important for ruling out similarly occurring lesions and in our case, except for one endodontically treated mandibular central incisor, all the other teeth were vital and unrestored, suggestive of focal OD. Most of the OD lesions are reported to be Periapical or focal lesions presenting as unilateral solitary lesions on the other hand, florid OD lesions are distributed symmetrically as multiple sclerotic masses, in one or more quadrants, usually in the tooth-bearing regions of the jaws.[13],[14],[18]

Majority of ODs are diagnosed during routine dental radiographic examination. Diagnosis becomes more challenging when a FOD lesion in its initial radiolucent stage is associated with an endodontically-treated tooth similar to our case. Advanced imaging (CT or MRI) is performed in such scenarios when conventional radiographs do not suggest definitive diagnosis &/or when there is a need for additional information to determine the extent of involvement, relationships with nearby anatomic structures, cortical expansion, internal density, etc.

OD requires no treatment of their benign nature and rarely produces symptoms. In an asymptomatic case, regular routine clinical checkups and periodic radiographic reassessments with follow-up orthopantamographic examination or Cone Beam Computed Tomography scans once in 2 or 3 years are recommended.[11],[19],[21]

The use of CT/ Cone Beam Computed Tomography scans serves as a valuable diagnostic tool particularly in symptomatic cases or during follow up.[20],[21],[22],[23],[24],[25] In this case, CBCT showed a hyper-dense image with mild resorption and expansion of cortical plates in the affected areas with clear and defined margins compatible with a FOL. A conclusive and definitive diagnosis is only possible with histopathologic examination of the biopsy sample that helps to differentiate OD/CODs from other similar lesions.[25],[26] An exploratory biopsy of the lesion was performed in our case to confirm the diagnosis of OD/COD that revealed cellular fibrous connective tissue mixed with osteoid trabaculae consisting of osteoblastic rimming and small irregular and round calcified areas suggestive of OD/COD.

FCOD can affect the diagnosis, management, and follow-up of clinical situations such as endodontic treatment, particulary with CODs that are increasingly radiodense obscuring the visibility of periodontal ligament and lamina dura rendering the endodontic procedure to rely completely on apex locators for apical information.[27] In cases that require periapical surgical intervention, compromised healing potential due to avascular nature of the bone cementum can make it susceptible to potential post-operative infection,due to reduced blood supply.[25],[26] In cases with concomitant periapical pathologies, determining the reduction in the size of the original lesion and its subsequent healing may not be easy.


  Conclusion Top


In view of these challenging features, it is clear that dentists should exercise basic knowledge about various lesions affecting the jaw bones to prevent misdiagnosis and avoid unnecessary treatments. It is important to understand the indications of endodontic treatment for its appropriate practical applicability for relevant cases. It is possible that during the initial consultation with the general dentist the lesion was probably in the initial osteolytic radiolucent stage mimicking periapical pathology leading to an unnecessary endodontic intervention. Repeated clinical examination, pulp sensitivity examination, gingival examination and radiographic imaging including Cone Beam Computed Tomography scans (2D and 3D imaging studies) are indicated to avoid misdiagnosis &/or mismanagement. Periapical osseous dysplasia should be considered when lesions occur in middle- aged women with unique or multiple radiolucent, mixed or radiopaque lesions in the tooth bearing areas of the jaw especially associated with asymptomatic and vital teeth.

Conflict of Interest: None



 
  References Top

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]



 

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