Florid Hypercementosis Synchronous with Periodontitis: a Case Report John K

RADIOLOGY/IMAGING Florid hypercementosis synchronous with periodontitis: a case report John K. Brooks, DDS/Ioana Ghita, DDS/Evan M. Vallee, BS/Adrien L. Charles-Marcel, DDS/ Jeffery B. Price, DDS, MS

Excessive cementum formation, referred to as hypercemento- ent the clinical, radiographic, and histopathologic findings of a sis (HC), is an uncommon nonneoplastic process that princi- 44-year-old female with moderate to severe periodontitis syn- pally occurs with permanent teeth. Widespread tooth involve- chronous with 22 HC-affected teeth. A list of other etiologies ment has been confined mostly to Paget disease of bone. Only associated with HC is provided. (Quintessence Int 2019;50: a limited number of reports of HC coincident with periodontitis 478–485; doi: 10.3290/j.qi.a42481) has appeared in the literature. The aim of this article is to pres-

Key words: etiology, florid, hypercementosis, periodontitis

Hypercementosis (HC) is characterized by an abnormal deposi- Case presentation tion of secondary radicular cementum, appearing as a spherical or irregularly shaped apical enlargement and typically found as An asymptomatic 44-year-old female sought comprehensive an incidental radiopaque finding.1 HC may be an isolated find- care at the University of Maryland School of Dentistry. The clin- ing or occur in more than one quadrant, predominately affect- ical examination was significant for generalized chronic, mod- ing posterior teeth.2 On rare occasions, HC may be symptom- erate to severe periodontitis and multiple caries lesions. Peri- atic and usually attributed to severe caries.3 The bulbous root odontal probing depths ranged from 2 to 7 mm. Tooth mobility configuration may pose increased difficulties when performing was restricted to the anterior teeth, mostly +2 grade, with only exodontia, orthodontic tooth movement, and endodontic ther- the maxillary right lateral incisor retained root tip (tooth 12 apy.4 In the earlier years of dentistry, some practitioners mistak- according to FDI notation) exhibiting a +3 mobility. HC was evi- enly believed that HC would promote an assortment of sys- dent radiographically with the maxillary right central incisor temic disorders, which prompted unnecessary extractions and and the left central and lateral incisor, canine, second premolar, the unfortunate persistence of the patient’s underlying illness.5 and third molar; the affected mandibular teeth were the left Although the primary cause for the majority of cases of HC first premolar, canine, and central incisor and the right canine, has been unclear, this dental malformation has been associated first premolar, and first molar (teeth 11, 21, 22, 23, 25, 28, 31, 33, with Paget disease of bone.6 There are only a scant number of 34, 43, 44, and 46) (Fig 1). The teeth with the most conspicuous published cases of HC concomitant with periodontitis.7-9 To involvement were 25, 28, 44, and 46 (Fig 2). Additionally, early increase awareness of their possible causal relationship, this signs of HC were seen with the maxillary right lateral incisor article describes the clinical, radiographic, and histopathologic and second premolar and the mandibular left lateral and right findings of a 44-year-old female with multiple teeth with HC and left central incisors (teeth 12, 15, 32, 41, and 42). Lesions and generalized chronic periodontitis. In addition, a list of consistent with focal osteosclerosis were visualized apical to other reported systemic, genetic, and dental comorbidities the mandibular left second premolar (tooth 35) and in the with HC is presented. edentulous left body of the mandible.

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1

Fig 1 Multiple hypercementosis-affected teeth evident in the intraoral complete radiographic series.

A panoramic radiograph taken 4.5 years previously was pro- bolic panel, ordered by the patient’s attending physician as cured and revealed five teeth with HC that had since been part of a routine physical examination, was obtained for ascer- extracted, namely the maxillary right first, second, and third tainment of HC and possible systemic correlation. These results molars and the maxillary left first and second molars (teeth 16, were significant for an elevated serum glucose of 164 mg/dl 17, 18, 26, and 27) (Fig 3). In total, the featured patient has had (normal 65 to 99 mg/dl) and vitamin D deficiency (25-hydroxy 22 teeth with radiographic manifestations of HC. It is unknown level 23.8 ng/ml [normal 30.0 to 100 ng/ml]). The serum calcium, whether any other previously extracted teeth displayed HC, as serum alkaline phosphatase, hepatic and renal function panel, the patient’s older dental records were not available. complete blood count, and platelets were within normal limits. The review of the medical history was salient for chronic The initial dental treatment plan consisted of extraction of all inactive hepatitis B, vitamin D deficiency, iron deficiency ane- remaining maxillary teeth (to be performed in separate quadrant mia, and headaches. Current medications included vitamin D, appointments) and construction of a conventional complete cyanocobalamin, and ibuprofen. A recent comprehensive meta- denture. All of the maxillary teeth were removed, with teeth 25

2a 2b 2c 2d

Figs 2a to 2d Intraoral periapical radiographs of prominent examples of hypercementosis. (a) Maxillary left second premolar. (b) Maxillary left third molar. (c) Mandibular right first premolar. (d) Mandibular right first molar.

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Fig 3 Panoramic radiograph taken 4.5 years previously revealed additional maxillary teeth with hypercementosis.

3

5

4b 4c Fig 5 Photomicrograph of the maxillary left third molar. Decalcified section showing Figs 4a to 4c Clinical views of two affected teeth. alternating layers of cellular and acellular (a) Maxillary left second premolar with adherent buccal plate. excessive cementum adjacent to root dentin (b) Maxillary left third molar (in toto). (hematoxylin and eosin stain, original 4a (c) Maxillary left third molar (hemisected, unstained). magnification ×10).

and 28 being particularly laborious to extract, owing to their root specimen demonstrated excessive deposition of cementum malformations. Extraction of tooth 25 was facilitated by the use consistent with HC. There was a prominent line of demarcation of a high-speed surgical handpiece to remove surrounding between the radicular tubular dentin and osteocementum. Of bone, although a segment of the buccal plate remained adher- interest, the radicular overgrowth appeared with alternating ent to the root (Fig 4a). Images of tooth 28 illustrate the exagger- zones of cellular (containing scattered cementocytes within the ated degree of radicular alteration (Figs 4b and 4c). lacunae) and acellular cementum (Fig 5). Due to the added surgical difficulty extracting the maxillary Postoperatively, the patient experienced only mild discom- left teeth, the patient was prescribed 500 mg amoxicillin (three fort. The patient is treatment planned to undergo extraction of times/day for 7 days) as a precaution for possible infection, multiple mandibular teeth, construction of a partial denture, chlorhexidine gluconate 0.12% antimicrobial rinse, and 600 mg and periodontal scaling. ibuprofen (four times/day as needed) for analgesia and as an anti-inflammatory agent. The gingiva overlying the maxillary left Discussion extraction sites was sutured with 3-0 chromic gut for wound approximation. HC is a relatively uncommon root anomaly and its incidence has Tooth 28 was placed in neutral buffered 10% formalin solu- ranged from 1.3% to 4.8% in various subpopulations (> 600 tion for fixation. Microscopic assessment of the decalcified patients), representing 0.12% to 0.96% of the total number of per-

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manent teeth examined.10-13 Conversely, far fewer instances of HC of these patients had synchronous HC. Other manifestations of have been recognized in the primary dentition.13 The mean age Paget disease include radiographically radiopaque/radiolucent of affected patients is 47.3 years5 and noted with a slight gender osseous lesions, bowing of the long bones, deep-seated bone predilection, occurring in 54.2% of females and 45.8% of males.9 pain, neurologic deficits, and markedly elevated serum alkaline Radiographically, HC appears as a homogenous radiopacity, phosphatase. Widespread HC was also reported in a study of slightly more radiolucent than dentin, expanding predominately 55% (55/100) of cohorts who ostensibly developed systemic along the apical root and infrequently encompassing most of the fluorosis from industrial pollution.22 Moreover, some nonsyn- radicular complex; the lamina dura and periodontal ligament dromic pedigrees of familial HC have been noted.4,23 This article space usually may be visualized encircling the cemental mass.14 provides the most comprehensive listing of HC associated with HC may range from localized to extensive involvement, with any systemic and genetic diseases, although nearly all of these one affected patient averaging 3.8 hypercementosed teeth.5 pathologies coincident with HC have been reported too infre- The oral pathology literature typically utilizes the descriptor quently to establish any definitive correlation (Table 1). florid for widespread or generalized cases of cemento-osseous There is widespread belief that subsets of cases of HC arise dysplasia. As such, the term “florid hypercementosis” has been in response to some low-grade oral inflammatory process, employed for the featured patient and it is advocated for particularly pulpal and periodontal disease, and owing in part patients with four or more affected teeth or having three or to microbial invasion.1,8 Through some ill-defined process, res- more affected quadrants. Overall, HC has been found in man- ident periodontopathogenic bacteria may induce cemental dibular teeth twice as often as maxillary teeth, notably with overproduction.8 Other oral etiologies of HC include ankylo- mandibular molars, less often with premolars, and least com- sis/infraocclusion, bruxism, sclerosing osteitis, hypodontia, monly with incisors and canines.9 occlusal traumatism, odontoma, physical trauma, simple Rarer variants of cementum overgrowth are coalescence of bone cyst, and supra-eruption (Table 2). Saad46 noted micro- roots of adjacent teeth (referred to as concrescence) or between scopic increases in cementum width in a small sample of the roots of an individual tooth; cementoblastoma; cementify- extracted primary mandibular second molars concurrent with ing fibroma; periapical, focal and florid cemento-osseous dys- agenesis of second premolars and suggested the cementum plasia; gigantiform cementoma; and formation of multiple hypertrophy may represent an adaptive process to prolonged cemental spikes.14-16 Sporadically, a cemental-like proliferation root anchorage. had been observed with titanium implants.17 Additional con- This featured article supports a tentative positive correla- siderations in the radiographic differential diagnosis of HC tion of HC with periodontitis. It is proposed that through some include exostosis, superimposed retained root tip, idiopathic undefined pathophysiologic mechanism, subsets of periodon- osteosclerosis, odontoma, intraosseous foreign body, osteoid tally involved teeth, pagetoid bone, and other comorbidities osteoma/osteoblastoma, and osteoma. associated with HC could promote cemental overgrowth as a In the majority of affected patients, cementum apposition compensatory response in susceptible hosts to plaque-induced represents an idiopathic process and is positively correlated alveolar bone loss (as seen in the featured patient), and perhaps with increased age.18 Specifically, in a study of 233 single-rooted comparable to the aforementioned pathogenesis theorized by teeth, cementum thickness averaged 0.076 mm from individu- Saad,46 thereby reducing the occurrence of tooth mobility via als younger than age 20 years old and increased to 0.215 mm increased root surface area. It is of interest that nearly 65 million for individuals from age 51 to 76 years old, nearly tripling in individuals in the United States have periodontitis52 and yet size.19 Azaz et al20 corroborated a similar cemental growth pat- only isolated cases of HC coincident with periodontitis have tern with age advancement in 23.3% (14/60) of impacted been published, either alone,7,8 or in combination with Pendred canines and premolars. syndrome.9 With the exception of Paget disease of bone and Although most publications have commonly reported an HC, the restricted number of recorded cases of periodontitis unspecified positive association of HC with Paget disease of and HC could reflect an aleatory relationship or be indicative of bone, elucidation of the actual incidence of this root malforma- underreporting, and additional case studies are warranted to tion within this subpopulation prompted the need to explore establish possible causation. Also left unresolved is whether the the historical dental literature. In 1938, Stafne and Austin21 featured patient’s vitamin D deficiency and potential reduction found only 16.7% (23/138) of patients with Paget disease had in bone density of the jaws could have played any stimulatory jawbone involvement, and rather demonstrably, 78.3% (18/23) role in the cementum overgrowth.

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Table 1 Disorders associated with hypercementosis

Etiology Other features Study

Acromegaly* Excessive skeletal growth, facies, macroglossia Kashyap et al24 Bisphosphonate-induced osteonecrosis Dental (pulpal necrosis, pulp stones, dentinoid/osteoid formations, ankyloses) de Camargo Moraes25 Calcinosis* Periarticular cystic and tumoral calcifications Burkes et al26 ENPPI mutation and generalized arterial Vascular calcifications, dental anomalies (infraocclusion, overretained primary Thumbigere-Math et al13 calcification of infancy* teeth, ankylosis, slow orthodontic tooth movement) Craniometaphyseal dysplasia* Hyperostosis of long bones and skull, palsies Martin et al27 Enamel dysplasia with odontogenic Unerupted teeth, hypodontia Peters et al28 fibroma-like hamartomas* Enamel-renal syndrome Enamel hypoplasia, renal dysfunction Wang et al29 Fluorosis† Osteopetrosis, periosteal bone and osteophyte formation, calcification of Teotia et al22 interosseous membrane and ligaments Gardner syndrome Osteomas, epidermoid cysts, skin fibromas, intestinal polyposis, supernumer- Arendt et al30 ary teeth Germodermia osteodysplastica* Thin and wrinkled skin, joint laxity, osteoporosis, bone fragility Lustmann et al31 Hyperparathyroidism-primary Generalized osseous demineralization, bone pain, lethargy, nausea, polyuria, Thoma32 arrhythmia, depression Hyperparathyroidism-secondary* Renal osteodystrophy Lopes et al33 Hyperthyoidism Goiter, exophthalmos, myxedema, heat intolerance, hyperhidrosis, cardiac Kupfer34 disease Hypoparathyroidism* Parathyroid hormone deficiency, hypocalcemia, hypophosphatemia Srirangarajan et al35 Inheritance* Isolated finding Zemsky23 Lobodontia* Misshapen teeth, hypodontia Ather et al36 Mucopolysaccharidosis type IV*‡ Skeletal deformities, short stature, enlarged organs, facies de Santana Sarmento et al37 Oculodentodigital syndrome* Anomalies (ocular, digital, teeth), facies Scheutzel38 Osteopetrosis* Severely dense bone, bone fragility, osteomyelitis García et al39 Paget disease* Osseous (pain, fractures, deformity, enlargement), neurologic deficits Rao and Karasick6 Pendred syndrome* Hearing loss, hypothyroidism Sharma and Pradeep9 Pyknodysostosis* Osteosclerosis, bone fragility, short stature, wide cranial sutures, brachyceph- Bathi and Masur40 aly, short fingers Rheumatic fever Streptococcal infection, cardiac valvulopathy, rash Neville et al41 Rheumatoid arthritis Joint (pain, deformation, destruction), fatigue Leider and Garbarino14 Sickle cell trait* Renal dysfunction, venothromboembolism Souza et al42 Systemic lupus erythematosus* Cutaneous and oral lesions, multiple organ system involvement Shoor et al43

*Published cases of affected patients with ≥ 3 teeth with hypercementosis. †55% of 100 cohorts identified with fluorosis and hypercementosis without specification of the number of affected teeth per individual. ‡7 affected teeth, (personal communication, Dmitry J. Sarmento, 30 September 2018).

In contradistinction, Bilgin et al53 noted reductions in cemen- HC almost invariably represents an asymptomatic radio- tal thickness in 92 periodontally involved extracted teeth follow- graphic discovery in an otherwise healthy tooth and does not ing root exposure to the oral environment due to attachment warrant treatment. Key features of HC are its indolent growth loss. Similarly, Paknejad et al54 conducted a pilot investigation behavior and absence of cortical expansion, pain, tenderness, involving eight sets of extractions from patients, aged 17 to 20 or paresthesia. Cases of atypical clinical or radiographic presen- years old, and demonstrated histologically diminished cemen- tation should undergo biopsy and submission for histopatho- tum width among all first molars with localized severe periodon- logic review. In situations where the affected tooth concur- titis, as compared to healthy third molars. These researchers sus- rently has pulpal/periodontal disease or is associated with pected that the cemental hypoplasia was consequent to the significant bruxism/occlusal traumatism, appropriate care periodontitis rather than as a promoter of the bone loss. should be focused on resolving the incident condition. When

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extracting teeth with HC, the attending clinician should con- Table 2 Dental etiologies associated with hypercementosis sider root sectioning and removal of contiguous bone to pre- vent adverse surgical sequelae, such as alveolar fracture, infec- Dental etiology Study tion, or the creation of a residual osseous defect. Kim et al55 Ankylosis/infraocclusion* Israel44 proposed use of intentional partial odontectomy in lieu of Bruxism Comuzzie and Steele45 complete extraction of mandibular molars with HC in close Hypodontia Saad46 proximity to the inferior alveolar nerve canal. Idiopathic Warrier and Vinayachandran,3 Basdra et al47 Furthermore, endodontic therapy on affected teeth may be Inflammation (pulpal or periodontal) Tadinada and Potluri1 complicated by an increased number of apical foramina and Occlusal traumatism Tadinada and Potluri1 the presence of resorptive defects.56 Improved outcomes might Odontoma and osseous dysplasia* Iida et al48 be achieved by the use of an apex locator for quantification of Periodontal disease Seed and Nixon,7 Zhou et al,8 the complex root canal system.57 Use of cone beam computed Sharma and Pradeep,9 present case* tomography may be helpful to distinguish HC from cemento- Physical trauma Prabhakar et al49 blastoma, based on the lack of alveolar expansion, as seen with Root fracture Tadinada and Potluri1 HC.58 It could also be anticipated that the completion of ortho- Root resorption Schätzle et al50 dontic movement of teeth with HC will take longer to achieve.3 Sclerosing osteitis Tadinada and Potluri1 Simple bone cyst Saito et al51 Supra-eruption* Present case Conclusions *Published cases of affected patients with ≥ 3 teeth with hypercementosis. This report has described an unusual case of extensive HC simultaneous with moderate to severe periodontitis. HC is a malformation of cemental hypertrophy and usually discovered as an incidental radiographic finding. Although its etiology is idiopathic in the vast majority of affected patients, the associa- tion of HC and Paget disease of bone is well known. It is posited that HC may arise secondary to periodontitis and possibly vita- min D deficiency in a subset of susceptible individuals as a Acknowledgments compensatory response to reduce tooth mobility. Additional published cases are warranted to establish clinicopathologic The authors thank Dmitry J. Sarmento, DDS, MS, Department of correlations. Dentistry, Paraíba State University, Araruna, Paraíba, Brazil, for providing the specific number of teeth with hypercementosis in one featured patient with mucopolysaccharidosis type IV Declaration (personal communication, 30 September 2018). In addition, the The documented clinical treatment conformed to the standard authors thank Mark Teske and Tom Jemski, The Photo and of care and was not performed for experimental purposes. The Graphics Group, University of Maryland School of Medicine, for authors declare no conflicts of interest. photographic expertise of the surgical specimens.

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John K. Brooks Ioana Ghita Resident/PhD Student, Graduate Program in Oral and Experimental Pathology, Department of Oncology and Diag- nostic Sciences, University of Maryland School of Dentistry, Balti- more, MD, USA.

Evan M. Vallee 3rd year predoctoral student, University of Mary- land School of Dentistry, Baltimore, MD, USA.

Adrien L. Charles-Marcel Intern, Department of Oral and Max- illofacial Surgery, University of Maryland School of Dentistry, Balti- more, MD, USA.

Jeffery B. Price Clinical Associate Professor, Director of Oral and John K. Brooks Clinical Professor, Department of Oncology and Maxillofacial Radiology, Department of Oncology and Diagnostic Diagnostic Sciences, University of Maryland School of Dentistry, Sciences, University of Maryland Dental School, Baltimore, MD, Baltimore, MD, USA. USA.

Correspondence: Dr John K. Brooks, Department of Oncology and Diagnostic Sciences, Room 7210, University of Maryland School of Dentistry, 650 West Baltimore Street, Baltimore, MD 21201, USA. Email: [email protected]

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