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Florid Cemento-osseous Dysplasia: A Case of Misdiagnosis
Case Report/Clinical Technique
Florid Cemento-osseous Dysplasia: A Case of Misdiagnosis Debora Delai, DDS, MSc,* Anarela Bernardi, DDS, MSc,* Gabriela Santos Felippe, DDS, MSc,* Cleonice da Silveira Teixeira, DDS, MSc, PhD,† Wilson Tadeu Felippe, DDS, MSc, PhD,† and Mara Cristina Santos Felippe, DDS, MSc, PhD† Abstract Florid cemento-osseous dysplasia (FCOD) belongs to the group of fibro-osseous lesions in which normal bone is replaced by fibrous connective tissue and calcified cementum tissue of the avascular type. Among the various types of fibro-osseous lesions, FCOD is 1 of the most commonly encountered diseases in clinical practice and may involve 3 or 4 of the quadrants. FCOD is located in the periapical regions of teeth, and the lesions are predominantly radiolucent (osteolytic phase), become mixed over time (cementoblast phase), and ultimately become radiopaque (osteogenic phase) with a thin radiolucent peripheral halo. The characteristics of FCOD in the initial stages are similar to those of periapical lesions of inflammatory origin, which may lead to misdiagnosis. A 38-year-old woman sought dental care because of complaints of pain on the right side of her face. A clinical examination revealed no marked alterations; a panoramic radiograph was therefore requested and revealed the presence of radiolucent lesions associated with the periapical regions of some of the lower teeth. Thus, the professional referred the patient for endodontic treatment of the associated teeth with the justification that the lesions were of endodontic origin. However, the endodontist found that the teeth responded positively to a sensitivity test. The initial diagnosis could have resulted in unnecessary root canal treatment, but after careful clinical, radiographic, and tomographic assessments by different professionals, FCOD was diagnosed, conservatively treated, and regularly monitored. It is important that dentists have a basic knowledge of the various injuries that affect the jaw bones to prevent errors in diagnosis and treatment and to promote oral health. (J Endod 2015;-:1–4)
T
Key Words
In June 2011, a 38-year-old woman sought dental care in a basic health unit of her city because of complaints of pain on the right side of her face, particularly in the morning. The patient was referred to the Center for Dental Specialties in Ararangua, Santa Catarina, Brazil, for evaluation by an oral surgeon. Because extra- and intraoral clinical examinations revealed no alterations, the oral surgeon requested a panoramic radiograph (Fig. 1) and a bilateral temporomandibular joint radiograph. The panoramic radiograph report indicated the ‘‘presence of a multilobular radiolucent image with
Differential diagnosis, florid cemento-osseous dysplasia, misdiagnosis, periapical lesion, pulp sensitivity test
he current classification of bone-related lesions launched in 2005 by the World Health Organization is based on sex; age; location; and the clinical, radiographic, and histopathological characteristics of the lesion. This classification includes cemento-ossifying fibromas, benign cementoblastomas, and benign fibro-osseous lesions (1). Among fibro-osseous lesions, cemento-osseous dysplasia is the most commonly found in clinical practice (2). Cemento-osseous dysplasia is characterized by the replacement of normal bone tissue with fibrous tissue that contains newly formed mineralized tissue (2, 3). Although its etiology is unknown, it is known that this lesion is benign (4) and originates from the proliferation of periodontal ligament cells (5). Cemento-osseous dysplasia is divided into 3 subtypes according to its clinical and radiographic features: periapical cemento-osseous dysplasia (PCOD), focal cementoosseous dysplasia, and florid cemento-osseous dysplasia (FCOD) (6). If the lesion affects 3 or 4 quadrants, it is generally considered to be FCOD (1). However, there is still controversy in the literature regarding the exact classification of the lesions (7–10). In severe cases, FCOD can result in bone expansion accompanied by pain and facial deformity (11). However, in most cases, the injuries are asymptomatic and can thus be diagnosed by routine radiographic examinations. When detected, a differential diagnosis that includes other bone lesions such as Paget disease, ossifying fibroma, and periapical lesions of inflammatory origin should be performed (12). Radiographically, the presence of lesions in the periapical regions of the teeth is observed; in the initial stages, these lesions are predominantly radiolucent (osteolytic phase), become mixed over time (cementoblast phase), and finally become radiopaque (osteogenic phase) with a thin radiolucent peripheral halo (2). Histologically, these lesions exhibit anastomosed trabecular bone and layers of calcification that are similar to cementum and are incorporated into a fibroblast matrix (13). Because of misdiagnoses, some cases of FCOD are referred to endodontic or even surgical treatment (14). This study describes the case of a patient with FCOD in which the lesions were initially confused with chronic inflammatory periapical lesions and later confused with odontomas or central osteoma. The aim of this case report was to highlight the clinical and radiographic features of FCOD to alert clinicians of the need to establish criteria for correct diagnosis and improved treatment.
Case Report
From the *Postgraduate Program and †Department of Endodontics, Federal University of Santa Catarina, Florianopolis, Santa Catarina, Brazil. Address requests for reprints to Dr Mara Cristina Santos Felippe, Department of Endodontics, School of Dentistry, Federal University of Santa Catarina, Florianopolis, Santa Catarina, Brazil. E-mail address: [email protected] 0099-2399/$ - see front matter Copyright ª 2015 American Association of Endodontists. http://dx.doi.org/10.1016/j.joen.2015.08.016
JOE — Volume -, Number -, - 2015
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Case Report/Clinical Technique
Figure 1. Panoramic radiograph (June 2011) showing the ‘‘presence of a multilobular radiolucent image with clear and regular contours located in the apical region of teeth #27, 28, and 29 compatible with odontogenic keratocysts or ameloblastoma.’’ Note the evidence of radiolucent foci (white arrows), foci with mixed radiopacity (blue arrows), and another radiopaque focus (red arrow) in the jaw.
Figure 3. Jaw CBCT scan (May 2013) showing ‘‘hypodense images surrounding the root apex of teeth #21, 22, 23, 24, 25, 26, 27, and 28 compatible with periapical fibrous dysplasia and a hyperdense image located on the alveolar bony ridge referring to tooth #31 (absent), with clear and defined limits, compatible with central osteoma or complex odontoma.’’ The CBCT scan reveals multiple foci of bone dysplasia in different stages of evolution in the periapical regions of the anterior and posterior teeth.
clear and regular contours located in the apical region of teeth #27, 28, and 29 compatible with odontogenic keratocysts or ameloblastoma.’’ The temporomandibular joint radiograph revealed an ‘‘impaction of the right mandibular head inside the mandibular fossa causing anatomical changes.’’ Based on these reports, the oral surgeon performed periapical radiography (Fig. 2A and B). Based on the analysis of the acquired images, the professional referred the patient to an endodontist to perform endodontic treatment of the associated teeth with the justification that the lesions were of endodontic origin. The endodontist found that teeth #27, 28, and 29 exhibited normal responses to pulp sensitivity and percussion tests. The patient was advised to return a few days later for reassessment by the oral surgeon. Because the symptoms disappeared, the patient only returned to the basic health unit in May 2013 with complaints of recurrence of the discomfort. Upon this return, the patient was seen by a periodontist who, based on a review of the data presented in medical records as well as the presence of symptoms and the absence of clinical signs, requested a jaw cone-beam computed tomographic (CBCT) scan (Fig. 3) to aid in the diagnosis. The CBCT scan indicated
the presence of hypodense areas surrounding the apices of the first premolars and most of the anterior teeth that were compatible with periapical fibrous dysplasia. Additionally, the CBCT scan indicated a hyperdense image in the alveolar bony ridge of tooth #31 (absent) with clear and defined limits that were compatible with a central osteoma or a complex odontoma. The case was then referred to the oral surgeon who, based on this information, indicated the need for surgery in the posterior mandible region to remove the supposed central osteoma or odontoma and referred the patient for histopathological examination. The lesion was not removed because, fortunately, another endodontist was consulted regarding the case. This endodontist studied the existing documentation and requested laboratory tests (ie, blood count, serum and urinary calcium, phosphorus, vitamin D, parathyroid hormone, and serum alkaline phosphatase levels) and a jaw CBCT scan to aid in the diagnosis. The laboratory tests indicated no alterations. The upper jaw CBCT scan provided evidence of ‘‘hypodense images located on the alveolar bony ridge of incisors and teeth #14 and 15 (visible in the cross section, not shown), consistent with PCOD, and mucosal
Figure 2. Periapical radiographs (June 2011) of (A) teeth #28 and 29 and (B) teeth #27 and 28. (A) shows a radiopaque focus with a radiolucent halo in the alveolar bony ridge related to tooth #31 (absent) (red arrow) and a radiolucent focus on the periapical region of tooth #28 (white arrow). (B) shows mixed radiopacity in the periapical region of tooth #27 (blue arrow).
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Figure 4. Upper jaw CBCT scan (July 2013). Coronal image (panoramic) indicating a possible mucous retention cyst in the left maxillary sinus (arrow).
thickening of the floor and side walls of the left maxillary sinus (Fig. 4), compatible with a mucous retention cyst’’. At that time (August 2013), the patient exhibited no pain symptoms. After careful clinical and radiographic evaluations and based on the CBCT and laboratory examination results, the lesions were diagnosed as FCOD. The painful symptoms were related to possible temporomandibular disorders or pathology of otolaryngology origin. The patient received explanations of FCOD, was informed of the importance of maintaining good oral hygiene, and was instructed to return every 6 months or upon the appearance of any sign or symptom. Based on the image that suggested a mucous cyst in the left maxillary sinus and the presence of temporomandibular joint pain, the patient was referred to an otolaryngology specialist and to a dentist specializing in temporomandibular disorders.
Discussion Cemento-osseous dysplasias comprise a group of disorders with the same origin and same pathological process but distinctive clinical and radiographic features (15). One of these dysplasias is PCOD, which is confined to the anterior region of the mandible (16), generally between the canines. FCOD is considered a widespread form of PCOD that affects 3 or 4 of the quadrants (6, 17, 18), as reported in this clinical case. The term ‘‘florid osseous dysplasia’’ was introduced by Melrose et al in 1976 (3). In 1985, Waldron (13) proposed the use of the term ‘‘FCOD’’ because the dense and sclerotic mass that forms resembles the cementum. FCOD is a benign and non-neoplastic disorder of unknown cause that consists of mesenchymal tissue fragments composed of spindle-shaped fibroblasts and collagen fibers with numerous small-caliber blood vessels. In this fibrous connective tissue, there is a mixture of trabecular bone, lamellar bone, and cementumlike particles (2). FCOD affects the bilateral maxillary teeth, and its incidence is higher among black women over 40 years of age (3); however, in the present clinical case, the patient was white and 38 years of age at the time of initial consultation. In this case, FCOD was initially diagnosed as inflammatory periapical disease, which is a fairly common mistake given the similarity of the radiographic aspects of the lesions when FCOD is in the osteolytic phase. If diagnosed by less attentive or less experienced professionals, the initial referral could have resulted in unnecessary root canal treatments. In this case, endodontic treatment was ruled out because the endodontist found that the teeth exhibited normal responses to pulp sensitivity and percussion tests. However, such JOE — Volume -, Number -, - 2015
normal responses may not always be present because in some clinical situations, such as teeth with large restorations, recent trauma, or atresia/calcification of the pulp chamber, the pulp may not respond to sensitivity tests. Thus, in addition to conducting a sensitivity test, professionals should also observe the integrities of the teeth (6) and search for evidence of pulp necrosis, such as caries, deep restorations, or dental trauma. In the cementoblast and osteogenic phases, the differential diagnosis of FCOD includes Paget disease (12, 15), diffuse chronic sclerosing osteomyelitis, cemento-ossifying fibroma, cementoblastoma, osteoblastoma, odontoma, hypercementosis (6, 19), and osteoma (13). Because the results of laboratory tests, including alkaline phosphatase levels, were normal, the possibility of Paget disease was ruled out; this conclusion was reached because in the active stages of this disease the blood alkaline phosphatase levels are elevated (3, 18, 20–22). Diffuse chronic sclerosing osteomyelitis should be considered a differential diagnosis only when an infectious process is directly responsible for bone sclerosis (10, 18, 23). In the present case, no related infection was detected. Other odontogenic tumors, particularly cemento-ossifying fibromas, typically exhibit greater buccolingual/palatal expansion than FCOD (24). The use of CBCT imaging has been described in the literature as a valuable diagnostic tool (8, 14, 20, 25). The 2013 CBTC report for the present case indicated a hyperdense image in the alveolar bony ridge of tooth #31 (absent) with clear and defined limits that were compatible with a central osteoma or complex odontoma. Osteomas are welldifferentiated, slow-growing lesions with radiopaque radiographic signatures that consist of mature bone tissue and typically do not cause pain symptoms (26). Odontomas are the most common benign tumors of odontogenic origin. According to the latest classification of the World Health Organization, there are 2 types of odontoma (ie, complex and compound). Complex odontomas are usually found in routine radiographs of the posterior maxillary region and are associated with an impacted tooth. Complex odontomas consist of radiopaque masses formed by enamel, dentin, cementum, and pulp that do not morphologically resemble teeth. Compound odontomas occur most commonly in the anterior maxillary region and consist of a collection of numerous small teeth (2). The CBTC examination of the patient also identified the presence of hypodense areas surrounding the root apical region of various other jaw teeth. In the maxilla, discrete hypodense foci were observed in the apical regions of teeth #14 and 15. Based on the locations of these lesions, epidemiological data, the results of the pulp sensitivity test, and the radiographic characteristics, this case was clinically diagnosed as FCOD. Importantly, after a definitive diagnosis of FCOD, there is often no need for any treatment of the lesion (27) because FCOD is a benign disease that rarely produces symptoms. However, even in the absence of signs and symptoms, reassessments with panoramic radiographs or CBCT scans every 2 or 3 years are indicated (8) to ensure that no changes occur in the benign features of the lesion (2, 28). Periodic follow-ups, prophylactic sessions, and hygiene instruction are also suitable for preventing the development of periodontal disease and tooth loss (8). Because the diagnosis of FCOD can be made based on clinical and radiographic examinations (20, 29), biopsies for histopathological analyses are not indicated because they may increase the risk of secondary infection or cause fractures in the surgical cavity region (8, 30). In cases with symptoms, surgical intervention is indicated (27) but can trigger secondary infection and induce osteomyelitis in the operated region (19). When inflammation or chronic infection occurs inside densely mineralized and avascular tissue with a low
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Case Report/Clinical Technique remodeling capacity (4), as in FCOD, there is a predisposition to the formation of ulcers on the mucosa and the possibility of exposure and bone loss (2, 31). Typically, antibiotic therapy is not effective for FCOD because the tissue diffusion of the drug is poor in this situation (8). In some clinical situations, FCOD can affect the diagnosis, treatment, and follow-up of teeth with endodontic involvement. When the lesion becomes more radiopaque, the periodontal ligament can become impossible to visualize radiographically, demanding the use of electronic apex locators to determine the working length (32). In cases of teeth with periapical disease, the reduction in the size of the original lesion and its subsequent healing may not be easily determined. In addition, if a periapical surgical intervention is required, the avascular nature of the bone cementumlike lesions and their subsequent compromised healing potential must be taken into consideration (32). In the present case, the patient was referred to an otolaryngology specialist and dentist specializing in temporomandibular disorders to investigate the origin of the pain. Since this referral was made, the patient has been regularly monitored through prophylactic sessions and provided with strict hygiene instructions; the FCOD remains asymptomatic. Given the previously described findings, it is clear that dentists should possess basic knowledge about various lesions that affect the jaw bones to prevent misdiagnoses and misleading treatments and to promote oral health.
Acknowledgments The authors deny any conflicts of interest related to this study.
References 1. Barnes L, Eveson JW, Reichart P, Sidransky D. World Health Organization Classification of Tumours: Pathology and Genetics of Head and Neck Tumours. Lyon, France: IARC Press; 2005. 2. Neville BW, Damm DD, Allen CM, Bouquot JE. Patologia Oral and Maxilofacial. Rio de Janeiro, RJ, Brazil: Guanabara Koogan; 2009. 3. Melrose RJ, Abrams AM, Mills BG. Florid osseous dysplasia: a clinical-pathological study of thirty-four cases. Oral Surg 1976;41:62–82. 4. G€und€uz K, Avsever H, Karac¸ayli U, et al. Florid cemento-osseous dysplasia: a case report. Braz Dent J 2009;20:347–50. 5. Kawai T, Hiranuma H, Kishino M, et al. Cemento-osseous dysplasia of the jaws in 54 Japanese patients: a radiographic study. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1999;87:107–14. 6. Komabayashi T, Zhu Q. Cemento-osseous dysplasia in an elderly Asian male: a case report. J Oral Sci 2011;53:117–20. 7. Waldron CA. Bone pathology. In: Neville BW, Damm DD, Allen CM, Bouquot JE, eds. Oral and Maxillofacial Pathology, 2nd ed. Philadelphia: Saunders; 2002: 533–87.
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8. Beylouni I, Farge P, Mazoyer JF, Coudert JL. Florid cemento-osseous dysplasia: report of a case documented with computed tomography and 3D imaging. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1998;85:707–11. 9. Kramer IR, Pindborg JJ, Shear M. The WHO histological typing of odontogenic tumours. A commentary on the second edition. Cancer 1992;70:2988–94. 10. MacDonald-Jankowski DS. Florid cemento-osseous dysplasia: a systematic review. Dentomaxillofac Radiol 2003;32:141–9. 11. Minhas G, Hodge T, Gill DS. Orthodontic treatment and cemento-osseous dysplasia: a case report. J Orthod 2008;35:90–5. 12. Mupparapu M, Singer SR, Milles M, Rinaggio J. Simultaneous presentation of focal cemento-osseous dysplasia and simple bone cyst of the mandible masquerading as a multilocularradiolucency. Dentomaxillofac Radiol 2005; 34:39–43. 13. Waldron CA. Fibro-osseous lesions of the jaws. J Oral Maxillofac Surg 1985;43: 249–62. 14. Escandarloo A, Yousefi F. CBTC findings of periapical cemento-osseous dysplasia: a case report. Imaging Sci Dent 2013;43:215–8. 15. Jerjes W, Banu B, Swinson B, Hopper C. Florid cemento-osseous dysplasia in a young Indian woman. A case report. Braz Dent J 2005;198:477–8. 16. Singer SR, Mupparapu M, Rinaggio J. Florid cemento-osseous dysplasia and chronic diffuse osteomyelitis: report of a simultaneous presentation and review of the literature. J Am Dent Assoc 2005;136:927–31. 17. Dagıstan S, Tozoglu €U, G€oregen M, C¸akur B. Florid cemento-osseous dysplasia: a case report. Med Oral Patol Oral Cir Bucal 2007;12:348–50. 18. Gonc¸alves M, Pıspico R, Alves FA, et al. Clinical, radiographic, biochemical and histological findings of florid cemento-osseous dysplasia and report of a case. Braz Dent J 2005;16:247–50. 19. Alsufyani NA, Lam EW. Osseous (cemento-osseous) dysplasia of the jaws: clinical and radiographic analysis. J Can Dent Assoc 2011;77:b70. 20. Ariji Y, Ariji E, Higuchi Y, et al. Florid cemento-osseous dysplasia: radiographic study with special emphasis on computed tomography. Oral Surg Oral Med Oral Pathol 1994;78:391–6. 21. Monti LM, Franc¸a DCC, Castro AL, et al. Displasia cemento-ossea florida: relato de caso. Odonto 2012;20:95–100. 22. Loh FC, Yeo JF. Florid osseous dysplasia in Orientals. Oral Surg Oral Med Oral Pathol 1989;68:748–53. 23. Araujo DB, Queiroz IV, Romero P, Falc~ao AFP. Displasia cemento-ossea florida. R Ci Med Biol 2005;4:167–73. 24. DelBalso A, Werning J. The role of computed tomography in the evaluation of cemento-osseous lesions. Oral Surg Oral Med Oral Pathol 1986;62:354–7. 25. Costa C, Haddad DS, Santos KCP, et al. Displasia cemento-ossea florida: relato de dois casos utilizando-se TCFC. Rev ABRO 2012;13:1. 26. Kaplan I, Calderon S, Buchner A. Peripheral osteoma of the mandible: a study of 10 new cases and analysis of the literature. J Oral Maxillofac Surg 1994;52: 467–70. 27. DiFiore P, Bowen S. Cemento-osseous dysplasia in African-American men: a report of two clinical cases. J Tenn Dent Assoc 2010;90:26–9. 28. Alves CR, Manzi FR. Displasia cemento-ossea florida: relato de caso clınico. Arq Bras Odontol 2007;3:25–31. 29. Gaetti-Jardim EC, Santiago Junior JF, Guastaldi FPS, et al. Displasia cemento-ossea florida: relato de caso. Rev Odontologica de Arac¸atuba 2010;31:31–4. 30. Summerlin DJ, Tomich CE. Focal cemento-osseous dysplasia: a clinicopathologic study of 221 cases. Oral Surg Oral Med Oral Pathol 1994;78:611–20. 31. Grasselli S, Da Silva CM, Fontanella VRC. Displasia ossea florida. Stomatos 2001;7: 55–8. 32. Tonioli MB, Schindler W. Treatment of a maxillary molar in a patient presenting with florid cemento-osseous dysplasia: a case report. J Endod 2004;30:665–7.
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Florid Cemento-osseous Dysplasia: A Case of Misdiagnosis Debora Delai, DDS, MSc,* Anarela Bernardi, DDS, MSc,* Gabriela Santos Felippe, DDS, MSc,* Cleonice da Silveira Teixeira, DDS, MSc, PhD,† Wilson Tadeu Felippe, DDS, MSc, PhD,† and Mara Cristina Santos Felippe, DDS, MSc, PhD† Abstract Florid cemento-osseous dysplasia (FCOD) belongs to the group of fibro-osseous lesions in which normal bone is replaced by fibrous connective tissue and calcified cementum tissue of the avascular type. Among the various types of fibro-osseous lesions, FCOD is 1 of the most commonly encountered diseases in clinical practice and may involve 3 or 4 of the quadrants. FCOD is located in the periapical regions of teeth, and the lesions are predominantly radiolucent (osteolytic phase), become mixed over time (cementoblast phase), and ultimately become radiopaque (osteogenic phase) with a thin radiolucent peripheral halo. The characteristics of FCOD in the initial stages are similar to those of periapical lesions of inflammatory origin, which may lead to misdiagnosis. A 38-year-old woman sought dental care because of complaints of pain on the right side of her face. A clinical examination revealed no marked alterations; a panoramic radiograph was therefore requested and revealed the presence of radiolucent lesions associated with the periapical regions of some of the lower teeth. Thus, the professional referred the patient for endodontic treatment of the associated teeth with the justification that the lesions were of endodontic origin. However, the endodontist found that the teeth responded positively to a sensitivity test. The initial diagnosis could have resulted in unnecessary root canal treatment, but after careful clinical, radiographic, and tomographic assessments by different professionals, FCOD was diagnosed, conservatively treated, and regularly monitored. It is important that dentists have a basic knowledge of the various injuries that affect the jaw bones to prevent errors in diagnosis and treatment and to promote oral health. (J Endod 2015;-:1–4)
T
Key Words
In June 2011, a 38-year-old woman sought dental care in a basic health unit of her city because of complaints of pain on the right side of her face, particularly in the morning. The patient was referred to the Center for Dental Specialties in Ararangua, Santa Catarina, Brazil, for evaluation by an oral surgeon. Because extra- and intraoral clinical examinations revealed no alterations, the oral surgeon requested a panoramic radiograph (Fig. 1) and a bilateral temporomandibular joint radiograph. The panoramic radiograph report indicated the ‘‘presence of a multilobular radiolucent image with
Differential diagnosis, florid cemento-osseous dysplasia, misdiagnosis, periapical lesion, pulp sensitivity test
he current classification of bone-related lesions launched in 2005 by the World Health Organization is based on sex; age; location; and the clinical, radiographic, and histopathological characteristics of the lesion. This classification includes cemento-ossifying fibromas, benign cementoblastomas, and benign fibro-osseous lesions (1). Among fibro-osseous lesions, cemento-osseous dysplasia is the most commonly found in clinical practice (2). Cemento-osseous dysplasia is characterized by the replacement of normal bone tissue with fibrous tissue that contains newly formed mineralized tissue (2, 3). Although its etiology is unknown, it is known that this lesion is benign (4) and originates from the proliferation of periodontal ligament cells (5). Cemento-osseous dysplasia is divided into 3 subtypes according to its clinical and radiographic features: periapical cemento-osseous dysplasia (PCOD), focal cementoosseous dysplasia, and florid cemento-osseous dysplasia (FCOD) (6). If the lesion affects 3 or 4 quadrants, it is generally considered to be FCOD (1). However, there is still controversy in the literature regarding the exact classification of the lesions (7–10). In severe cases, FCOD can result in bone expansion accompanied by pain and facial deformity (11). However, in most cases, the injuries are asymptomatic and can thus be diagnosed by routine radiographic examinations. When detected, a differential diagnosis that includes other bone lesions such as Paget disease, ossifying fibroma, and periapical lesions of inflammatory origin should be performed (12). Radiographically, the presence of lesions in the periapical regions of the teeth is observed; in the initial stages, these lesions are predominantly radiolucent (osteolytic phase), become mixed over time (cementoblast phase), and finally become radiopaque (osteogenic phase) with a thin radiolucent peripheral halo (2). Histologically, these lesions exhibit anastomosed trabecular bone and layers of calcification that are similar to cementum and are incorporated into a fibroblast matrix (13). Because of misdiagnoses, some cases of FCOD are referred to endodontic or even surgical treatment (14). This study describes the case of a patient with FCOD in which the lesions were initially confused with chronic inflammatory periapical lesions and later confused with odontomas or central osteoma. The aim of this case report was to highlight the clinical and radiographic features of FCOD to alert clinicians of the need to establish criteria for correct diagnosis and improved treatment.
Case Report
From the *Postgraduate Program and †Department of Endodontics, Federal University of Santa Catarina, Florianopolis, Santa Catarina, Brazil. Address requests for reprints to Dr Mara Cristina Santos Felippe, Department of Endodontics, School of Dentistry, Federal University of Santa Catarina, Florianopolis, Santa Catarina, Brazil. E-mail address: [email protected] 0099-2399/$ - see front matter Copyright ª 2015 American Association of Endodontists. http://dx.doi.org/10.1016/j.joen.2015.08.016
JOE — Volume -, Number -, - 2015
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Figure 1. Panoramic radiograph (June 2011) showing the ‘‘presence of a multilobular radiolucent image with clear and regular contours located in the apical region of teeth #27, 28, and 29 compatible with odontogenic keratocysts or ameloblastoma.’’ Note the evidence of radiolucent foci (white arrows), foci with mixed radiopacity (blue arrows), and another radiopaque focus (red arrow) in the jaw.
Figure 3. Jaw CBCT scan (May 2013) showing ‘‘hypodense images surrounding the root apex of teeth #21, 22, 23, 24, 25, 26, 27, and 28 compatible with periapical fibrous dysplasia and a hyperdense image located on the alveolar bony ridge referring to tooth #31 (absent), with clear and defined limits, compatible with central osteoma or complex odontoma.’’ The CBCT scan reveals multiple foci of bone dysplasia in different stages of evolution in the periapical regions of the anterior and posterior teeth.
clear and regular contours located in the apical region of teeth #27, 28, and 29 compatible with odontogenic keratocysts or ameloblastoma.’’ The temporomandibular joint radiograph revealed an ‘‘impaction of the right mandibular head inside the mandibular fossa causing anatomical changes.’’ Based on these reports, the oral surgeon performed periapical radiography (Fig. 2A and B). Based on the analysis of the acquired images, the professional referred the patient to an endodontist to perform endodontic treatment of the associated teeth with the justification that the lesions were of endodontic origin. The endodontist found that teeth #27, 28, and 29 exhibited normal responses to pulp sensitivity and percussion tests. The patient was advised to return a few days later for reassessment by the oral surgeon. Because the symptoms disappeared, the patient only returned to the basic health unit in May 2013 with complaints of recurrence of the discomfort. Upon this return, the patient was seen by a periodontist who, based on a review of the data presented in medical records as well as the presence of symptoms and the absence of clinical signs, requested a jaw cone-beam computed tomographic (CBCT) scan (Fig. 3) to aid in the diagnosis. The CBCT scan indicated
the presence of hypodense areas surrounding the apices of the first premolars and most of the anterior teeth that were compatible with periapical fibrous dysplasia. Additionally, the CBCT scan indicated a hyperdense image in the alveolar bony ridge of tooth #31 (absent) with clear and defined limits that were compatible with a central osteoma or a complex odontoma. The case was then referred to the oral surgeon who, based on this information, indicated the need for surgery in the posterior mandible region to remove the supposed central osteoma or odontoma and referred the patient for histopathological examination. The lesion was not removed because, fortunately, another endodontist was consulted regarding the case. This endodontist studied the existing documentation and requested laboratory tests (ie, blood count, serum and urinary calcium, phosphorus, vitamin D, parathyroid hormone, and serum alkaline phosphatase levels) and a jaw CBCT scan to aid in the diagnosis. The laboratory tests indicated no alterations. The upper jaw CBCT scan provided evidence of ‘‘hypodense images located on the alveolar bony ridge of incisors and teeth #14 and 15 (visible in the cross section, not shown), consistent with PCOD, and mucosal
Figure 2. Periapical radiographs (June 2011) of (A) teeth #28 and 29 and (B) teeth #27 and 28. (A) shows a radiopaque focus with a radiolucent halo in the alveolar bony ridge related to tooth #31 (absent) (red arrow) and a radiolucent focus on the periapical region of tooth #28 (white arrow). (B) shows mixed radiopacity in the periapical region of tooth #27 (blue arrow).
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Figure 4. Upper jaw CBCT scan (July 2013). Coronal image (panoramic) indicating a possible mucous retention cyst in the left maxillary sinus (arrow).
thickening of the floor and side walls of the left maxillary sinus (Fig. 4), compatible with a mucous retention cyst’’. At that time (August 2013), the patient exhibited no pain symptoms. After careful clinical and radiographic evaluations and based on the CBCT and laboratory examination results, the lesions were diagnosed as FCOD. The painful symptoms were related to possible temporomandibular disorders or pathology of otolaryngology origin. The patient received explanations of FCOD, was informed of the importance of maintaining good oral hygiene, and was instructed to return every 6 months or upon the appearance of any sign or symptom. Based on the image that suggested a mucous cyst in the left maxillary sinus and the presence of temporomandibular joint pain, the patient was referred to an otolaryngology specialist and to a dentist specializing in temporomandibular disorders.
Discussion Cemento-osseous dysplasias comprise a group of disorders with the same origin and same pathological process but distinctive clinical and radiographic features (15). One of these dysplasias is PCOD, which is confined to the anterior region of the mandible (16), generally between the canines. FCOD is considered a widespread form of PCOD that affects 3 or 4 of the quadrants (6, 17, 18), as reported in this clinical case. The term ‘‘florid osseous dysplasia’’ was introduced by Melrose et al in 1976 (3). In 1985, Waldron (13) proposed the use of the term ‘‘FCOD’’ because the dense and sclerotic mass that forms resembles the cementum. FCOD is a benign and non-neoplastic disorder of unknown cause that consists of mesenchymal tissue fragments composed of spindle-shaped fibroblasts and collagen fibers with numerous small-caliber blood vessels. In this fibrous connective tissue, there is a mixture of trabecular bone, lamellar bone, and cementumlike particles (2). FCOD affects the bilateral maxillary teeth, and its incidence is higher among black women over 40 years of age (3); however, in the present clinical case, the patient was white and 38 years of age at the time of initial consultation. In this case, FCOD was initially diagnosed as inflammatory periapical disease, which is a fairly common mistake given the similarity of the radiographic aspects of the lesions when FCOD is in the osteolytic phase. If diagnosed by less attentive or less experienced professionals, the initial referral could have resulted in unnecessary root canal treatments. In this case, endodontic treatment was ruled out because the endodontist found that the teeth exhibited normal responses to pulp sensitivity and percussion tests. However, such JOE — Volume -, Number -, - 2015
normal responses may not always be present because in some clinical situations, such as teeth with large restorations, recent trauma, or atresia/calcification of the pulp chamber, the pulp may not respond to sensitivity tests. Thus, in addition to conducting a sensitivity test, professionals should also observe the integrities of the teeth (6) and search for evidence of pulp necrosis, such as caries, deep restorations, or dental trauma. In the cementoblast and osteogenic phases, the differential diagnosis of FCOD includes Paget disease (12, 15), diffuse chronic sclerosing osteomyelitis, cemento-ossifying fibroma, cementoblastoma, osteoblastoma, odontoma, hypercementosis (6, 19), and osteoma (13). Because the results of laboratory tests, including alkaline phosphatase levels, were normal, the possibility of Paget disease was ruled out; this conclusion was reached because in the active stages of this disease the blood alkaline phosphatase levels are elevated (3, 18, 20–22). Diffuse chronic sclerosing osteomyelitis should be considered a differential diagnosis only when an infectious process is directly responsible for bone sclerosis (10, 18, 23). In the present case, no related infection was detected. Other odontogenic tumors, particularly cemento-ossifying fibromas, typically exhibit greater buccolingual/palatal expansion than FCOD (24). The use of CBCT imaging has been described in the literature as a valuable diagnostic tool (8, 14, 20, 25). The 2013 CBTC report for the present case indicated a hyperdense image in the alveolar bony ridge of tooth #31 (absent) with clear and defined limits that were compatible with a central osteoma or complex odontoma. Osteomas are welldifferentiated, slow-growing lesions with radiopaque radiographic signatures that consist of mature bone tissue and typically do not cause pain symptoms (26). Odontomas are the most common benign tumors of odontogenic origin. According to the latest classification of the World Health Organization, there are 2 types of odontoma (ie, complex and compound). Complex odontomas are usually found in routine radiographs of the posterior maxillary region and are associated with an impacted tooth. Complex odontomas consist of radiopaque masses formed by enamel, dentin, cementum, and pulp that do not morphologically resemble teeth. Compound odontomas occur most commonly in the anterior maxillary region and consist of a collection of numerous small teeth (2). The CBTC examination of the patient also identified the presence of hypodense areas surrounding the root apical region of various other jaw teeth. In the maxilla, discrete hypodense foci were observed in the apical regions of teeth #14 and 15. Based on the locations of these lesions, epidemiological data, the results of the pulp sensitivity test, and the radiographic characteristics, this case was clinically diagnosed as FCOD. Importantly, after a definitive diagnosis of FCOD, there is often no need for any treatment of the lesion (27) because FCOD is a benign disease that rarely produces symptoms. However, even in the absence of signs and symptoms, reassessments with panoramic radiographs or CBCT scans every 2 or 3 years are indicated (8) to ensure that no changes occur in the benign features of the lesion (2, 28). Periodic follow-ups, prophylactic sessions, and hygiene instruction are also suitable for preventing the development of periodontal disease and tooth loss (8). Because the diagnosis of FCOD can be made based on clinical and radiographic examinations (20, 29), biopsies for histopathological analyses are not indicated because they may increase the risk of secondary infection or cause fractures in the surgical cavity region (8, 30). In cases with symptoms, surgical intervention is indicated (27) but can trigger secondary infection and induce osteomyelitis in the operated region (19). When inflammation or chronic infection occurs inside densely mineralized and avascular tissue with a low
Florid Cemento-osseous Dysplasia
3
Case Report/Clinical Technique remodeling capacity (4), as in FCOD, there is a predisposition to the formation of ulcers on the mucosa and the possibility of exposure and bone loss (2, 31). Typically, antibiotic therapy is not effective for FCOD because the tissue diffusion of the drug is poor in this situation (8). In some clinical situations, FCOD can affect the diagnosis, treatment, and follow-up of teeth with endodontic involvement. When the lesion becomes more radiopaque, the periodontal ligament can become impossible to visualize radiographically, demanding the use of electronic apex locators to determine the working length (32). In cases of teeth with periapical disease, the reduction in the size of the original lesion and its subsequent healing may not be easily determined. In addition, if a periapical surgical intervention is required, the avascular nature of the bone cementumlike lesions and their subsequent compromised healing potential must be taken into consideration (32). In the present case, the patient was referred to an otolaryngology specialist and dentist specializing in temporomandibular disorders to investigate the origin of the pain. Since this referral was made, the patient has been regularly monitored through prophylactic sessions and provided with strict hygiene instructions; the FCOD remains asymptomatic. Given the previously described findings, it is clear that dentists should possess basic knowledge about various lesions that affect the jaw bones to prevent misdiagnoses and misleading treatments and to promote oral health.
Acknowledgments The authors deny any conflicts of interest related to this study.
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