|Year : 2015 | Volume
| Issue : 1 | Page : 19-24
Florid osseous dysplasia with Klebsiella associated osteomyelitis and periapical osseous dysplasia in Asian females: A report of two cases
Sinny Goel1, Aarti Singh1, Anju Garg2, Sujoy Ghosh1, Shikha Gupta1, Sunita Gupta1
1 Department of Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi, India
2 Department of Radiodiagnosis, Lok Nayak Jaiprakash Hospital, New Delhi, India
|Date of Web Publication||18-Feb-2015|
Dr. Sinny Goel
Oral Medicine and Radiology, Maulana Azad Institute of Dental Sciences, New Delhi
Source of Support: None, Conflict of Interest: None
Periapical osseous dysplasia (POD) and florid osseous dysplasia (FOD) are benign jaw conditions with normal architecture of bone replaced by fibroblast, collagen fibers and mineralized material. We are presenting a case of FOD with Klebsiella infection that rarely infects the jaws and a case of POD with rare maxillary involvement and role of cone beam computed tomography (CBCT) in its diagnosis. Evaluation with conventional and higher imaging was done. Pus culture was done for FOD associated infection. The case with POD was followed-up for 2 years to differentiate it from FOD. Case with extensive multiquadrant swellings and discharging sinus was diagnosed as FOD with Klebsiella infection and minor surgery performed successfully under antibiotic coverage. Another case was diagnosed as POD with CBCT, which revealed maxillary lesions not seen in two-dimensional imaging. Diagnosis was confirmed after a long follow-up due to its limited extent. Elective surgery can be performed under antibiotic coverage in FOD. POD can be seen in maxilla, and CBCT and long follow-up have important role in diagnosis.
Keywords: klebsiella , osseous dysplasia, osteomyelitis
|How to cite this article:|
Goel S, Singh A, Garg A, Ghosh S, Gupta S, Gupta S. Florid osseous dysplasia with Klebsiella associated osteomyelitis and periapical osseous dysplasia in Asian females: A report of two cases. J Oral Maxillofac Radiol 2015;3:19-24
|How to cite this URL:|
Goel S, Singh A, Garg A, Ghosh S, Gupta S, Gupta S. Florid osseous dysplasia with Klebsiella associated osteomyelitis and periapical osseous dysplasia in Asian females: A report of two cases. J Oral Maxillofac Radiol [serial online] 2015 [cited 2019 Sep 17];3:19-24. Available from: http://www.joomr.org/text.asp?2015/3/1/19/151644
| Introduction|| |
The latest World Health Organization's classification (WHO) in 2005 of benign tumors recognized florid osseous dysplasias (FOD) and periapical osseous dysplasias (POD) as lesions associated with bone and designated them as FOD and POD that form along with focal osseous dysplasia (OD) and familial gigantiform cementoma, the group of OD. 
Florid osseous dysplasia is an OD first described by Melrose et al. in 1976.  It belongs to the spectrum of fibro-osseous lesions and represents a reactive process in which normal bone is replaced by cellular fibrous connective tissues and poorly cellularized mineralized material.  FOD is commonly seen in women of middle age (40-50 years old). It often occurs bilaterally in the mandible with symmetric involvements,  but it can also affect the maxilla. The condition may be totally asymptomatic and in such cases, the lesion is detected when radiographs are taken for some other purposes. However, it may become symptomatic due to a secondary infection, and it becomes difficult to treat once infected due to avascularity of the areas affected with FOD. 
Periapical osseous dysplasia occurs most commonly in the anterior mandible with a female predilection. Although cases have been reported with both mandible as well as maxillary lesions, the maxillary involvement is uncommon. , Condition is asymptomatic, and the involved teeth are vital as in FOD. The diagnosis can be made on the basis of the appropriate radiological and clinical characteristics. ,
This paper describes the cases diagnosed as FOD and POD on the basis of clinical and radiological findings with OD seen on histopathological examination. The case diagnosed as FOD was complicated by Klebsiella infection that very rarely infects the bones, especially the jaw bones. The case of multifocal POD is presented with maxillary-mandibular involvement, and importance of cone beam computed tomography (CBCT) is demonstrated to evaluate the lesions in three-dimensions.
| Case Reports|| |
A 64-year-old female reported to the outpatient department of tertiary care center with a discharging sinus in the right submental region present since last 2 years and swelling associated with dull pain in anterior mandibular edentulous ridge region since last 3-4 years, which has been progressively increasing in size. Although the patient noticed a gradual increase in size of both the jaws since last 4 years, but more of the lower jaw. Extraoral examination showed facial asymmetry due to the swelling of the right and left mandibular body region and discharge of purulent exudate from the sinus opening surrounded by the erythematous skin. Swelling was irregular, ill-defined, bony hard, nodular and nontender [[Figure 1]a]. Intraoral examination revealed obliteration of lower left buccal vestibule and swollen lingual soft tissue on the right side. Exposed dome shaped smooth yellowish mass (2.5 cm × 2 cm) was present over right mandibular anterior ridge area and was tender, bony hard, fixed and it was easily removed and probably was a calculus deposit. Irregular brownish exposed bone on ridge area was seen anterior to the carious maxillary right first molar with displaced maxillary right canine [[Figure 1]b]. Based on the clinical findings of expansion of the jaws, the diagnosis was initially suspected to be Paget's disease.
|Figure 1: (a) Extraoral view revealing facial asymmetry and sinus opening in right submental region, (b) intraoral view revealing swollen edentulous ridge and yellowish mass present anteriorly|
Click here to view
Orthopantomogram [Figure 2] revealed multiple impacted maxillary and mandibular teeth surrounded by globular, irregular radiopacities with irregular radiolucent border and extending to the inferior alveolar nerve region. Similar opacities were seen in the periapical zone of erupted teeth not attached to the root. Lesions extended till the superior margin of mandible with respect to no. 35 and no. 36, which appeared to be displaced occlusally. Based on this multiquadrant finding of radiopaque lesions, limited to the dentate areas the case was provisionally diagnosed as FOD. Computed tomography (CT) scan confirmed the presence of mixed-density, expansile lesion with areas of sclerosis and ground-glass attenuation involving bilateral maxilla and mandible with areas of bony erosion, representative of FOD [[Figure 3]a and b]. Well-defined hyperdense calcific lesion was noted within the left frontal sinus extending into the anterior ethmoid sinuses causing irregularity of the medial wall of the orbit, a separate finding possibly of ivory osteoma [[Figure 3]c].
|Figure 2: Orthopantomogram shows irregular radiopaque globular masses involving all the four quadrants|
Click here to view
|Figure 3: (a) Non contrast computed tomography (NCCT) mandible axial view revealing globular radiopaque areas in the mandible body region (black empty arrow) with ground-glass appearance of the surrounding bone (white arrow), (b) NCCT maxilla axial view revealing globular radiopaque areas present in maxillary alveolus bilaterally (arrows), (c) NCCT skull axial view revealing dense osteoma like radiopacity apparent in the left ethmoid sinus (arrow)|
Click here to view
Serum calcium, phosphate and alkaline phosphatase levels were within normal limits. Also, there was no systemic skeletal abnormality seen when radiographs were taken for other regions. On pus culture and sensitivity testing, Klebsiella was isolated, and patient was subjected to a short course of antibiotics, which resulted in complete disappearance of pus discharge and healing of sinus opening. An incisional biopsy was taken from the mandibular and maxillary areas of expansion. The histopathological examination revealed dysplastic osseous tissue with resting and reversal lines [Figure 4]. These findings although not specific of FOD, are suggestive of OD.
|Figure 4: Sclerotic mass of osseous material containing few marrow spaces. Resting and reversal lines throughout the osseous tissue are suggestive of dysplastic osseous tissue|
Click here to view
A 37-year-old female patient reported to the outpatient department of a tertiary care center with the chief complaint of swelling in mandibular right posterior region since 10-15 days. On examination, slight dome shaped expansion of buccal cortex was seen on the right side of mandible in no. 46, no. 47 region [Figure 5]. It was hard and nontender, and the overlying mucosa was normal in color. Associated teeth were vital on electric pulp testing. Blood chemistry was found to be within normal limits.
|Figure 5: Intraotal view revealing slight expansion of buccal cortex in #46, #47 region|
Click here to view
Orthopantomogram revealed multiple periapical radiolucent lesions extending interdentally, with sclerosed margins and central radiopacity in mandible in the anterior and posterior regions and maxillary anterior region [Figure 6]. Based on these findings, a provisional diagnosis of POD was made. Cone beam computed tomography scan was advised to enable detailed visualization of the lesion in multiple planes as in panoramic view a lesion was appearing to arise from the root of no. 46, which is a case with cementoblastoma, although it usually presents as a solitary lesion without any radiolucent stage in its development. CBCT images [Figure 7] revealed periapical radiolucent areas extending interdentally, with sclerosed well-defined margins and central homogeneous radiopacity, present in canine, premolar and molar regions of the mandible bilaterally; having similar appearance as periapical enodontic lesions; however, an association of simple bone cyst scalloping around the roots of no. 36 and no. 37 cannot be ruled out [[Figure 7]b]. Associated teeth were vital helping in the differentiation from an endodontic lesion. A similar lesion was seen in maxillary incisor region without a central radiopacity. Similar CBCT findings were seen after 2 years of follow-up, with only slightly increased opacity of the central radiopaque areas. Thus, the bilaterally symmetric presence of the lesions along with small size; with no change in the size over a long term confirmed the diagnosis of POD.
|Figure 6: Orthopantomogram shows multifocal lesions present bilaterally extending from mesial side of the canine till distal side of third molar|
Click here to view
|Figure 7: Cone beam computed tomography (CBCT) images revealing periapical osseous dysplasia. (a) Reformatted panoramic CBCT image shows multiple lesions present bilaterally extending from mesial side of the canine till distal side of third molar (b) sagittal view shows a well-defined periapical radiolucency scalloping around the roots of no. 36, no. 37 (associated simple bone cyst), with sclerosed margin and central homogeneous radiopacity (c) coronal CBCT shows well defined periapical radiolucencies with sclerosed margins in molar regions bilaterally with central homogeneous radiopacities not attached to root and expansion of right buccal cortex (arrow) (d) axial CBCT image shows the radiolucent lesion around the apical section of the right maxillary incisor root (white arrow) and discontinuity of the patatal cortex (e) three-dimensional reconstruction view depicting the labial aspect of the same lesion as in Figure 7d|
Click here to view
| Discussion|| |
Osseous dysplasias are a group of nonneoplastic fibro-osseous lesions; ,, characterized by an alteration of bone structure and its replacement by fibroblasts, collagen fibers and mineralized material.  OD represents a spectrum of conditions. However, for the purposes of current study, OD was considered to represent two main conditions: POD, defined as a condition related to the one or more anterior or posterior teeth; and FOD, defined as a condition with more extensive involvement (at least 2 quadrants). 
Florid osseous dysplasia was first described by Melrose et al. (1976).  The terminology and classification of these fibro-osseous lesions has long been a matter of discussion for pathologists and clinicians. , The second edition of the WHOs Histological Typing of Odontogenic tumors in 1992 recognized them as florid cemento-osseous dysplasia (FCOD), that form with periapical cemental dysplasia and other COD, the group of COD.  Because of the debate during the last decades about; whether the cementum-like tissue is present, it has been decided to give-up the term cement in the latest WHO's classification of odontogenic tumors in 2005. COD has, therefore, been called OD.  This classification recognizes in OD 4 groups;  POD, which occurs in the periapical area of the teeth in the anterior region of the mandible ,, and sometimes in maxilla;  FOD, a more extensive lesion; focal OD; and familial gigantiform cementoma. ,
The true incidence of the FOD is unknown and at present, there is no satisfactory explanation for the reported gender and racial predilection.  The epidemiology of POD is similar to that of FOD.  FOD and POD are most commonly seen in the middle aged (40-50) women, , which may suggest that sex-linked factors are implicated in the etiology. However, etiology is unknown.  Histopathology reveals that these may be due to reactive or dysplastic changes of the periodontal ligament (PDL),  but PDL spaces of the related teeth are visible radiographically, so there seems a little possibility of origin from PDL. 
Osseous dysplasias mimics radiographic endodontic periapical pathosis in early radiolucent stages.  Without proper pulp testing and diagnosis, the radiographic presentation in the periapical area could easily be misdiagnosed as periapical periodontitis. In our case 2, the pulp of associated teeth was found to be vital with electric pulp test and thus the case was considered to be a mixed-stage POD, which generally requires no treatment. Infection may result in the formation of drainage tracts intraorally or extraorally,  as in our case of FOD.
The radiographic appearance of FOD and POD lesions varies depending upon the stage.  The lesion is poorly circumscribed and radiolucent initially  and opacifies progressively with deposition of the mineralized tissue.  The same lesion may appear in different stages at 1-time. The classic appearance of FOD, as in case 1, includes diffuse, lobular, irregular radiopacities in dentate regions of the alveolar process of maxilla and mandible. , POD and FOD lesions generally have a limited growth potential,  but FOD cases may show extensive involvement and expansion.  The size of the FOD lesions as in our case, can vary from <1 cm to 10 cm,  while POD lesions are limited to a size of 1-2 cm, with more of mesiodistal diameter than superoinferior usually,  as in our case; where no change in the size of initial small lesions was seen even after a long follow-up. The location of the lesions is usually confined to the inter-radicular and periapical area, although an aggressive lesion of FOD may expand more inferiorly and superiorly. , In the mandible, lesions do not involve the inferior border and rami, and they are found superior to the inferior alveolar nerve.  Finally, CT scanning or CBCT can be useful in the evaluation of extension of the lesions and their relationship with the neighboring structures, in particular in the maxilla.  The radiopacities in the case 2, appearing to be attached to the root in panoramic view was seen in multiplanar CBCT images to be away from the root. This suggests the osseous origin of the lesions of POD speculating the role of CBCT in their diagnosis. Also, a maxillary lesion, which was not seen in two-dimensional imaging was well appreciated in CBCT axial images, which though is an uncommon site for POD. 
These clinical and radiographic features of FOD and POD may suggest some other pathologies that are important to differentiate from these; such as FOD should be differentiated from diffuse sclerosing osteomyelitis;  characterized by symptoms of inflammation with varying degrees of sclerosis or lucency.  In the mixed stage and radiopaque stage, the differential diagnosis of POD might include chronic sclerosing osteomyelitis and odontoma; as these lesions exhibit similar internal calcifications as POD.  POD is usually detected on conventional radiological examinations; however, higher imaging is required to differentiate from other similar appearing lesions. High-density mass in POD that is centered in the low-density area is different from the findings of calcifying cystic odontogenic tumor in which calcification is observed at or near the cyst wall.  Scalloping of the radiolucency associated with the lesions may suggest the unusual association of simple bone cyst with the OD,  as seen in [Figure 7]b.
The histopathological feature of FOD is similar to all the other three types of OD including POD. It depends on the stage or the degree of calcification of the lesion. Early lesions are composed of cellular fibrous tissue containing variable amounts of new woven bone trabeculae with osteoblast rimming. Occasional spherical calcifications may also be seen. In more mature lesions, there may be woven and lamellar bone and sclerotic masses of globular basophilic calcifications. There may be prominent reversal and resting lines giving a pagetoid appearance, , as seen in our case.
Ours was a case of Klebsiella osteomyelitis complicating a case of FOD. Klebsiella is a member of the Klebsiella genus of Enterobacteriaceae family and belongs to the normal flora of the human mouth and intestine.  There are only a few cases reported with Klebsiella osteomyelitis in bony skeleton. In a study by Kharbanda and Dhir of 110 patients with osteomyelitis; the organisms isolated were staphylococci (40%), streptococci (4%), Klebsiella (4%), proteus (6%) and mixed organisms (30%).  However, there are no cases reported till now in the jaw and in association with FOD, unlike in our case of Klebsiella osteomyelitis associated with FOD. Biopsy and multiple extractions were done under antibiotic coverage without any complications until 6 months of the follow-up period.
Regarding the treatment of POD and FOD in case of asymptomatic lesions, treatment and biopsy are not indicated. It is wise to keep the patient under observation. ,,, In the absence of clinical signs, revaluation with panoramic radiographs every 2 or 3 years is adequate.  The treatment must also be preventive to avoid infectious complications provoked by untimely tooth extractions, biopsies and trauma of covering fibromucosa; due to decreased vascularity of lesions. ,, An antibiotic prophylaxis is also recommended prior to endodontic treatment or any other dental care. , Infection may not respond to antibiotics due to poor tissue diffusion;  however, unexpectedly in our case antibiotics have shown favorable results with a resolution of pain and discharging sinus opening.
| Conclusion|| |
We conclude that elective surgical procedures can be performed successfully under antibiotic coverage in FOD. POD lesions, though rarely; can be seen in maxilla, and CBCT and long follow-up have an important role in the evaluation of the extent and origin, and hence the diagnosis of these lesions.
| References|| |
Barnes L, Eveson JW, Reichart PA, Sidransky D. Pathology and Genetics of Head and Neck Tumours. World Health Organization Classification of Tumours. Lyon: IARC Press; 2005. p. 284.
Melrose RJ, Abrams AM, Mills BG. Florid osseous dysplasia. A clinical-pathologic study of thirty-four cases. Oral Surg Oral Med Oral Pathol 1976;41:62-82.
Lin TM, Huang WH, Chiang CP, Lin HN, Liao YS, Chiang ML. Florid cemento-osseous dysplasia (FCOD): Case report. J Dent Sci 2010;5:242-5.
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.
Falace DA, Cunningham CJ. Periapical cemental dysplasia: Simultaneous occurrence in multiple maxillary and mandibular teeth. J Endod 1984;10:455-6.
Komabayashi T, Zhu Q. Cemento-osseous dysplasia in an elderly Asian male: A case report. J Oral Sci 2011;53:117-20.
Kramer IR, Pindborg JJ, Shear M. 2nd ed. Berlin: Springer; 1992. Histological typing of odontogenic tumours.
Heuberger BM, Bornstein MM, Reichart PA, Hürlimann S, Kuttenberger JJ. Dysplasie osseuse periapicale anterieure du maxillaire superieur. Rev Mens Suisse Odontostomatol 2010;120:1007-11.
Su L, Weathers DR, Waldron CA. Distinguishing features of focal cemento-osseous dysplasias and cemento-ossifying fibromas: I. A pathologic spectrum of 316 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:301-9.
Alsufyani NA, Lam EW. Osseous (cemento-osseous) dysplasia of the jaws: Clinical and radiographic analysis. J Can Dent Assoc 2011;77:b70.
MacDonald-Jankowski DS. Florid cemento-osseous dysplasia: A systematic review. Dentomaxillofac Radiol 2003;32:141-9.
Slootweg PJ. Bone diseases of the jaws. Int J Dent 2010;2010:702314.
Kawai T, Hiranuma H, Kishino M, Jikko A, Sakuda M. 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.
Young SK, Markowitz NR, Sullivan S, Seale TW, Hirschi R. Familial gigantiform cementoma: Classification and presentation of a large pedigree. Oral Surg Oral Med Oral Pathol 1989;68:740-7.
Resnick CM, Novelline RA. Cemento-osseous dysplasia, a radiological mimic of periapical dental abscess. Emerg Radiol 2008;15:367-74.
Bencharit S, Schardt-Sacco D, Zuniga JR, Minsley GE. Surgical and prosthodontic rehabilitation for a patient with aggressive florid cemento-osseous dysplasia: A clinical report. J Prosthet Dent 2003;90:220-4.
Coleman H, Altini M, Kieser J, Nissenbaum M. Familial florid cemento-osseous dysplasia - A case report and review of the literature. J Dent Assoc S Afr 1996;51:766-70.
Kakehashi S, Stanley HR, Fitzgerald RJ. The effects of surgical exposures of dental pulps in germ-free and conventional laboratory rats. Oral Surg Oral Med Oral Pathol 1965;20:340-9.
Marx RE, Stern D. Oral and Maxillofacial Pathology: A Rationale for Diagnosis and Treatment. 1 st
ed. Illinois: Quintessence Publishing; 2003.
Benjelloun L, EL Harti K, EL Wady W. Florid osseous dysplasia: Report of two cases and a review of the literature. Int J Odontostomatol 2011;5:257-66.
Ackermann GL, Altini M. The cementomas - A clinicopathological re-appraisal. J Dent Assoc S Afr 1992;47:187-94.
Köse TE, Köse OD, Karabas HC, Erdem TL, Ozcan I. Findings of florid cemento-osseous dysplasia: A report of three cases. J Oral Maxillofac Res 2013;4:e4.
Eskandarloo A, Yousefi F. CBCT findings of periapical cemento-osseous dysplasia: A case report. Imaging Sci Dent 2013;43:215-8.
Gonçalves M, Píspico R, Alves Fde A, Lugão CE, Gonçalves A. Clinical, radiographic, biochemical and histological findings of florid cemento-osseous dysplasia and report of a case. Braz Dent J 2005;16:247-50.
Groot RH, van Merkesteyn JP, Bras J. Diffuse sclerosing osteomyelitis and florid osseous dysplasia. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1996;81:333-42.
Ariji Y, Ariji E, Higuchi Y, Kubo S, Nakayama E, Kanda S. Florid cemento-osseous dysplasia. Radiographic study with special emphasis on computed tomography. Oral Surg Oral Med Oral Pathol 1994;78:391-6.
White SC, Pharoah MJ. Oral Radiology - Principles and Interpretation. 4 th
ed. Saint Louis: Mosby; 2000.
Speight PM, Carlos R. Maxillofacial fibro-osseous lesions. Curr Diagn Pathol 2006;12:1-10.
Fang CT, Lai SY, Yi WC, Hsueh PR, Liu KL, Chang SC. Klebsiella pneumoniae
genotype K1: An emerging pathogen that causes septic ocular or central nervous system complications from pyogenic liver abscess. Clin Infect Dis 2007;45:284-93.
Kharbanda Y, Dhir RS. Natural course of hematogenous pyogenic osteomyelitis (a retrospective study of 110 cases). J Postgrad Med 1991;37:69-75.
Bayi EH, El Harti K, El Wady W. Dysplasie cementaire periapicale. Inf Dent 2004;86:2527-30.
Sadda RS, Phelan J. Dental management of florid cemento-osseous dysplosia. N Y State Dent J 2014;80:24-6.
Ali S, Nankoo S, Syed AB, Yang J. Florid osseous dysplasia with superimposed mandibular osteomyelitis. Skeletal Radiol 2014;43:679, 709-10.
Dumas M, Ohanian H, Forest D. La dysplasie cementoosseuse floride. J Dent Que 2000;37:97-101.
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]