Infected Cemento-Osseous Dysplasia: Analysis of 66 Cases and Literature Review.

The aim of this study was to describe a series with 66 cases of infected cemento-osseous dysplasia (COD) and to discuss the demographic distribution, clinicoradiographic features and treatment of this condition. A study looking back on the diagnoses made at a single Brazilian centre within a 28-year timeframe was performed. A literature review with searches across five databases was also conducted to identify reports on osteomyelitis/infected COD. Descriptive and statistical analyses were performed. The case series study showed a female/male ratio of 21:1. Affected individuals' mean age was 57.4 years. Mandible was the most affected site (95.5%) and florid subtype was the most frequent infected COD (62.1%). Tooth extraction was the main factor associated with the development of infection associated within a COD lesion. The literature review retrieved 30 studies reporting 46 cases of this condition. Asian women in their 40 s and 50 s were more affected. Surgery for removal/curettage of necrotic bone was acknowledged as an appropriate approach to the treatment of this infection. The clinicodemographic data of the study were similar to data collected across the literature. Clinicians, maxillofacial surgeons and oral rehabilitation providers should be alert to the diagnosis of COD, since infection is a frequent complication whose management is challenging.

Peripheral Cemento-Ossifying Fibroma withUncommon Clinical Presentation: A Case Report / Fibroma periférico cemento-ossificante con una presentación clínica poco común: Reporte de caso

Abstract Peripheral cemento-ossifying fibroma (PCOF) is a reactive gingival over growth, occurring frequently in the maxillary anterior region of teenage and young females. Peripheral cemento-ossifying fibroma (PCOF) is supposed to be originating from periosteum and/or periodontal ligament. A large number of factors have been implicated in the pathogenesis of PCOF, which includes trauma, local irritation, calculus and hormonal disturbances. The definitive diagnosis of PCOF is based upon its clinical, radiological and histological features. Because of the high recurrence rate (8-20%) of PCOF, a close post-operative follow-up is required. Herewith, we are presenting a case of PCOF in 24-year old female patient at an uncommon location.

Resumen El fibroma cemento-osificante periférico (PCOF, por sus siglas en inglés) es un crecimiento gingival reactivo, que se produce con frecuencia en la región anterior del maxilar en adolescentes y mujeres jóvenes. Se supone que el fibroma cemento-osificante periférico (PCOF) se origina en el periostio y/o el ligamento periodontal. Una gran cantidad de factores han sido implicados en la patogénesis de PCOF, dentro de las que se incluye trauma, irritación local, cálculo y alteraciones hormonales. El diagnóstico definitivo de PCOF se basa en sus características clínicas, radiológicas e histológicas. Debido a la alta tasa de recurrencia (8-20%) , se requiere un seguimiento post-operatorio estricto. Mediante este artículo presentamos un caso de PCOF en una paciente de 24 años de edad y el manejo teraupético.

Familial Gigantiform Cementoma: Case Report of an Unusual Clinical Manifestation and Possible Mechanism Related To "Calcium Steal Disorder".

Familial gigantiform cementoma is an exceedingly rare but distinct subtype of cemento-osseous-fibrous lesion. Undocumented radiographic changes and related bone metabolism disorder are herein hypothesized and discussed. We present an adolescent case with recurrent familial gigantiform cementoma who received surgical intervention in our hospital. Apart from typical multiquadrant and expansile abnormalies involving both jaws, he also suffered from several times of fractures in lower extremity. Furthermore, radiographic examinations of calvaria, pelvis, femoris, tibia, and fibula all revealed radiolucent areas signifying diffuse osteopenic bone losses. Some of his consanguineous relatives bore the same burden of fractures during pubertal period.Considering these polyostotic conditions, a correlation of congenital bone metabolism disorder in cases with familial gigantiform cementoma, named "calcium steal disorder," was thus proposed. Familial gigantiform cementoma is closely associated with "calcium steal disorder." Whole-body dual-energy absorptiometry should be considered as a routine examination for fracture-related risk prediction.

Secondary jaw aneurysmal bone cyst (JABC)--a possible misnomer? A review of literature on secondary JABCs, their pathogenesis and oncogenesis.

CONTEXT: Aneurysmal bone cysts are rare pseudocysts, commonly seen in long bones and vertebral column. Although a well described and reported lesion, many misconceptions still prevail regarding their etiopathogenesis. Many of the reported cases of jaw aneurysmal bone cysts (JABC) present with another bone pathology. AIMS: The purpose of this review was to evaluate the incidence of neoplastic lesions occurring simultaneously with a JABC (in contrast to primary JABCs). Any pathogenetic and oncogenetic association between primary and secondary jaw ABCs has been reviewed and discussed. SETTINGS AND DESIGN: A methodical narrative review of literature was performed, given the incidence of mostly case reports on this topic. METHODS AND MATERIAL: A methodical electronic search of Pubmed, Pubmed Central, Medline and Cochrane databases was performed for reported cases of JABC. These articles were analysed and segregated into primary and secondary ABC and, if secondary, the lesion it concurrently occurred with. Another search was conducted to yield articles discussing the cytopathogenetic and oncogenetic origins of ABCs. RESULTS AND CONCLUSIONS: About 15% of the ABCs reported were of secondary nature. Amongst the associated lesions, cement-ossifying fibroma and ossifying fibroma were the most common, followed by fibrous dysplasia and central giant cell granuloma. No ABCs were associated with metastatic changes. The search for histopathogenesis pointed to a specific cytogenetic abnormality as the origin of primary ABCs, with USP6 as its main oncogene and spindle cell as the neoplastic cell, unlike with secondary ABCs, suggesting that they are distinct pathological processes.

Clinicopathologic conference: multiquadrant expansile fibro-osseous lesion in a juvenile.

This paper intends to present clinical, radiologic, and histopathologic features of a case of gigantiform cementoma manifesting in a 14-year-old boy, without any familial involvement. He has been followed for 3 years. Further cases from the literature have been reviewed that confirm a similar clinicopathologic picture. This is the first case report that describes "non-familial gigantiform cementoma" after the World Health Organisation's classification of 2005.

Osseous (cemento-osseous) dysplasia of the jaws: clinical and radiographic analysis.

OBJECTIVES: To explore the demographic characteristics and clinical features among patients with osseous (cemento-osseous) dysplasia (OD/COD) of the jaws and to determine the frequency of particular radiographic characteristics. METHODS: The charts and radiographic reports of 118 patients with OD/COD, obtained from the archives of the University of Toronto discipline of oral and maxillofacial radiology, were reviewed. Demographic and clinical data, radiographic findings and final diagnoses were collected and analyzed to determine typical characteristics. RESULTS: Of the 117 patients for whom age and sex were known, the majority (97 [82.9%]) were female; these female patients had a mean age (± standard deviation) of 44.3 ± 13.4 years. Eighty-three (72.2%) of the 115 patients for whom symptoms were known were clinically asymptomatic. Ninety-three patients (78.8%) had OD/COD at single sites (i.e., periapical OD/COD), and 25 (21.2%) had OD/COD at multiple sites (i.e., florid OD/COD). In addition, 15 (12.7%) of the cases were associated with one or more simple bone cysts, and 13 (11.0%) were associated with osteomyelitis. In most cases, the OD/COD was unilateral, with the lesion being located in the mandible, usually associated only with the posterior teeth. The lesions exhibited well-defined, sclerotic or corticated margins (108 patients [91.5%]) and were surrounded by a radiolucent border. Minimal effects on surrounding structures were observed. As well, 85 (72.0%) of the lesions were in the mixed radiolucent-radiopaque stage, with dense, cementum-like radiopacities. CONCLUSIONS: The majority of cases of OD/COD occurred in women in the fifth decade of life, and most cases were asymptomatic. OD/COD was more likely to present as solitary lesions but also occurred in association with simple bone cysts or osteomyelitis.

Clinical and radiographic features of solitary and cemento-osseous dysplasia-associated simple bone cysts.

UNLABELLED: The simple bone cyst (SBC) is a pseudocyst that can occur as a solitary entity in the jaws or may occur in association with cemento-osseous dysplasia (COD). OBJECTIVE: The purpose of this study was to review the clinical and radiographic features of solitary and COD-associated SBCs. METHODS: Archived imaging reports from the Special Procedures Clinic in Oral and Maxillofacial Radiology at the Faculty of Dentistry at the University of Toronto between 1 January 1989 and 31 December 2009 revealed 23 COD-associated SBCs and 68 solitary SBCs. RESULTS: Almost all solitary and COD-associated SBCs were found in the mandible. Furthermore, 87.0% of COD-associated SBCs were found in females in their fifth decade of life (P < 0.001) while solitary SBCs were found in equal numbers in both sexes in their second decade of life (P < 0.005). COD-associated SBCs were also more likely to cause thinning of the endosteal cortex, bone expansion and scalloping of the superior border between teeth (all P < 0.001) than solitary SBCs that are classically described as having these characteristics. Finally, COD-associated SBC demonstrated a loss of lamina dura more often (P < 0.05) than solitary SBCs. CONCLUSIONS: Knowledge of the sporadic association between COD and SBC and their potential radiographic appearances should prevent inappropriate treatment and management of these patients.

[Long-term follow-up of cemento-osseous dysplasia: a contralateral recurrence before radiotherapy]. / Évolution à long terme de la dysplasie cémento-osseuse: une récidive controlatérale avant radiothérapie.

INTRODUCTION: Cemento-osseous dysplasia is a benign fibro-osseous lesion of the jaws. Its complications are very rare. OBSERVATION: A panoramic x-ray was made to complete the pre-radiotherapy oral assessment of a 71-year-old female patient presenting with lung adenocarcinoma. This revealed a periapical fibro-osseous lesion on tooth n° 46. She had presented with a similar lesion on tooth n° 36, 16 years before. This had been treated by extraction and alveolar curettage. No surgical treatment was suggested for this recurrence on tooth n° 46. DISCUSSION: The diagnosis of focal cemento-osseous dysplasia is usually made on radiological and clinical data. The risk for transition to a florid form is unknown. Exeresis surgery before radiotherapy is discussed.

The orthodontic management of a patient presenting with cemento-osseous dysplasia.

Cemento-osseous dysplasias are sometimes seen on routine radiographic examination of the tooth-bearing areas of the jaws, most commonly amongst adult female patients. Lesions present as radiolucent to radio-opaque areas, depending on various levels of maturity. Extraction of teeth is strongly contraindicated because there is a high risk of infection with a possibility of bone sequestration, as antibiotics are unable to cross the thick cortical marginal bone that surrounds these lesions. No report has been found in the literature on the orthodontic management of patients with this condition. This paper reports on a 28-year-old black female patient with cemento-osseous dysplasia who received orthodontic treatment.

Florid cemento-osseous dysplasia: a case report.

Florid cemento-osseous dysplasia (FCOD) has been described as a condition that characteristically affects the jaws of middle-aged black women. It usually exhibits as multiple radiopaque cemetum-like masses distributed throughout the jaws. Radiographically, FCOD appears as dense, lobulated masses, often symmetrically located in various regions of the jaws. Computed tomography, because of its ability to give axial, sagittal, and frontal views, is useful in the evaluation of these lesions. This paper presents the case of a patient who was diagnosed with FCOD on the basis of clinical and radiographic findings.

Florid cemento-osseous dysplasia: a case report

Florid cemento-osseous dysplasia (FCOD) has been described as a condition that characteristically affects the jaws of middle-aged black women. It usually exhibits as multiple radiopaque cemetum-like masses distributed throughout the jaws. Radiographically, FCOD appears as dense, lobulated masses, often symmetrically located in various regions of the jaws. Computed tomography, because of its ability to give axial, sagittal, and frontal views, is useful in the evaluation of these lesions. This paper presents the case of a patient who was diagnosed with FCOD on the basis of clinical and radiographic findings.

A displasia cemento-óssea florida (DCOF) tem sido descrita como uma condição que afeta tipicamente os maxilares de mulheres negras de meia idade, geralmente exibindo massas radiopacas semelhantes ao cemento, distribuídas nos ossos maxilares. Radiograficamente, a DCOF apresenta-se como densas masas lobuladas, frequentemente distribuídas simetricamente em diversas regiões dos maxilares. A tomografia computadorizada, que oferece vistas axial, sagital e frontal, é útil na avaliação dessas lesões. Este artigo apresenta o caso de uma paciente em que a DCOF foi diagnoasticada com base nos achados clínicos e radiográficos.

Orthodontic treatment and cemento-osseous dysplasia: a case report.

This report presents a case of florid cemento-osseous dysplasia (FCOD), affecting the mandibular teeth, in a 23-year-old Somalian female patient for whom orthodontic treatment was undertaken. The presenting features of the patient and the effect of orthodontic treatment on this case are discussed.

Dental findings in a family with hyperparathyroidism-jaw tumor syndrome and a novel HRPT2 gene mutation.

Hyperparathyroidism-jaw tumor syndrome (HPT-JT) is an important diagnosis because of the possible involvement of other family members and risk of malignant disease. We report clinical and genetic studies in a previously undocumented Australian family with HPT-JT. The proband and his sister presented with bilateral or recurrent mandibular radiolucencies diagnosed histopathologically as cemento-ossifying fibromas. Mutation screening of the recently identified disease gene HRPT2 was performed by direct sequencing in 3 affected members. This revealed a novel mutation in exon 1 of HRPT2 (nt 20AGGACG --> GGGAG), which is predicted to inactivate the parafibromin protein through protein truncation and premature termination of translation. The terminology used for the jaw lesions in this syndrome warrants review to become more consistent. Cemento-ossifying fibroma is the preferred term to better reflect the pathologies found in most individuals and families,and to emphasize the significance of the jaw lesions in the diagnosis of the syndrome.

Simultaneous presentation of focal cemento-osseous dysplasia and simple bone cyst of the mandible masquerading as a multilocular radiolucency.

The fibro-osseous lesions represent a large group of disorders that have many common characteristics including clinical, radiographic and microscopic features. Although most are of unknown aetiology, some are believed to be neoplastic and others are related to metabolic imbalances. It is not unusual to see these lesions presenting with a range of radiographic appearances, causing considerable diagnostic confusion owing to their similar histology. This case report presents a histologically benign cemento-osseous lesion that is radiographically consistent with a focal cemento-osseous dysplasia, along with a concomitant simple (traumatic) bone cyst within the right mandibular alveolus, with the initial presentation as a multilocular radiolucency. Pertinent literature is reviewed.

Familial gigantiform cementoma with brittle bone disease, pathologic fractures, and osteosarcoma: a possible explanation of an ancient mystery.

We describe four individuals of an African-American family with a predominantly diaphyseal bone disease associated with familial gigantiform cementoma (FGC), a disorder typically seen in Caucasians. The mother and her children presented with deformities of the jaws, abnormalities of the long bones, and pre-pubertal pathologic fractures. The index patient carried the diagnosis of osteosarcoma (OS). In addition, we provide a possible explanation for the jaw abnormalities of King Tutankhamen's father in the 18th dynasty in Egypt around 1350 BC.