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. 2011 Jul;40(5):282–289. doi: 10.1259/dmfr/32556437

Fibrous dysplasia—a 13-year retrospective radiographic analysis in a south Indian population

V Nityasri 1,*, PS Haris 2, T Bose 1, A Balan 1
PMCID: PMC3520263  PMID: 21697153

Abstract

Objective

The aim of this study was to examine the clinical and radiographic presentation of fibrous dysplasia through a 13-year retrospective study in patients who reported to the outpatient unit of Government Dental College, Trivandrum.

Methods

The clinical file records and radiographs of the patients who reported to the outpatient clinic in the Department of Oral Medicine and Radiology were retrospectively reviewed for histopathologically diagnosed fibrous dysplasia. A detailed analysis of the clinical and radiographic features of the 24 cases retrieved was carried out.

Results

The male-to-female ratio of incidence was approximately 1:1. Most of the patients were in the second or third decade of life. Almost all the patients presented with a complaint of swelling on the side of the face. The mandible was more frequently involved than the maxilla. The most common radiographic pattern observed was the “ground-glass” appearance. Most of the patients exhibited expansion of the involved bone and loss of lamina dura of associated teeth.

Conclusion

The awareness of protean features of fibrous dysplasia evident through this study is essential in the accurate diagnosis and proper treatment planning of such lesions.

Keywords: fibrous dysplasia, ground glass, craniofacial, jaw

Introduction

Fibrous dysplasia is a perplexing disease of bone, of unknown aetiology, uncertain pathogenesis and diverse histology.1,2 It is often described as a hamartomatous malformation characterized by idiopathic arrest in the normal maturation of bone in the woven bone stage, interspersed with abnormal proliferation of fibrous connective tissue containing varying amounts of osteoid and osseous structures of presumably metaplastic nature.3 The term fibrous dysplasia was first used by Lichtenstein in 1938.4 Post-zygotic mutation in the guanine nucleotide-binding protein, α-stimulating activity polypeptide 1 (GNAS 1) gene is the recently proposed aetiology for fibrous dysplasia. It has been suggested that clinical severity of the lesion depends on the time of appearance of the mutation.5,6 There are two primary categories of the disease: monostotic fibrous dysplasia that involves only one bone and polyostotic fibrous dysplasia, which involves several bones. A monostotic form does not progress into a polyostotic form of the disease.7 Jaffe–Lichtenstein syndrome is a variant of polyostotic fibrous dysplasia with café-au-lait pigmentation of the skin. A more severe form of the polyostotic form of fibrous dysplasia accompanied by endocrine disturbances of varying types in addition to skin pigmentation is called the McCune–Albright syndrome. Another category identified as the craniofacial type of fibrous dysplasia is confined to the face and jaws involving two or more bones.8

Fibrous dysplasia creates radiographic patterns that are virtually indistinguishable from other lesions affecting the bones, such as Paget's disease and cemento-osseous fibroma. Difficulty is often experienced when the lesion is detected on radiographs with negligible clinical evidence. Awareness is required regarding the myriad of radiographic appearances on account of its local effects on the jaw, and also because of the possibility of extensive lesions of this entity in the skeleton.

In order to obtain a better appreciation of the clinical presentation and the intriguing radiographic features of fibrous dysplasia, all the cases of this disease which came to the outpatient department of Government Dental College, Trivandrum, during the period of 1996–2010 were analysed retrospectively.

Materials and methods

The clinical record library of the Department of Oral Medicine and Maxillofacial Radiology, Government Dental College, Trivandrum, a tertiary referral centre, was retrospectively reviewed and all the histopathologically diagnosed cases of fibrous dysplasia documented from November 1996 until February 2010 were analysed. This period selected is coincident with the tenure of the current head of department and the file records she instituted. The Institutional Ethical Committee approval was not required as this was a retrospective study based on file records of patients who gave informed consent prior to documentation and treatment.

Patients were not included in this series if adequate histories and relevant radiographic data could not be obtained. The features were entered into a pro forma, highlighting the radiological features. Conventional plain film radiographs such as panoramic radiographs and intraoral radiographs were available for all cases supplemented with other skull views. Intraoral periapical radiographs were considered for all cases at the suspected peripheries of the lesions in order to define the exact extent of the lesion. CT was generally performed when the borders of the lesions could not be discerned from plain radiographs, especially in the lesions affecting the anatomically complex maxilla.

The various study variables considered were age, sex, chief complaint and its duration, type of fibrous dysplasia and the site of the lesion in jaw bones. The various radiographic parameters studied were the periphery of the lesion; internal structure; presence of areas of cystic degeneration; effects on adjacent structures, including displacement of inferior alveolar canal; encroachment into paranasal sinuses; expansion of the lower border of the mandible; thinning or loss of cortex; loss of lamina dura; and root resorption. The accepted radiological criteria from literature and standard text books such as White and Pharaoh and Worth were considered.9,10 The definition of boundaries was according to the criteria set by Slootweg and Muller.11 A lesion was considered to be well demarcated when its radiodensity changed significantly within a distance of 1 mm when passing to the surrounding bone.

Results

Patient demographics

During the 13 year period of study, 24 cases were histopathologically diagnosed as fibrous dysplasia. Three cases which were identified as fibro-osseous lesions with doubtful histological pictures and inadequate radiographic data were not included in this study. The cases were in the age range of 9–66 years at presentation with an average age of 29.75 years. An increased prevalence of the disease was seen in the second and third decade of life. Male patients were more affected, with 13 cases, while females accounted for 11 cases; a male-to-female ratio of incidence being approximately 1.18:1. The chief complaint of 21 patients was an aesthetically disfiguring swelling of the face. The older age group of patients primarily presented with pain of odontogenic origin of short duration associated with a barely detectable swelling. The mean age of the patients when prior awareness of the complaint was considered was calculated to be 19.2 years. 18 cases were classified as having monostotic lesions, 5 cases had craniofacial lesions and 1 case had a polyostotic presentation.

Anatomic location of lesions

Among the cases of fibrous dysplasia affecting facial bones, the mandible was reported as the most affected bone in 13 cases, of which 11 cases solely involved the mandible (Table 1). Maxillary involvement was seen in 12 of the cases, of which 7 cases involved the maxilla alone (Table 2). Four cases involving the zygoma and four involving the frontal and temporal bone were considered as the craniofacial form of fibrous dysplasia (Figure 1, Table 3). In the single case of polyostotic fibrous dysplasia, apart from the jaws, the ethmoid, occipital bone, the femur and the ribs were also involved (Table 3). The unilateral presentation of fibrous dysplasia was apparent in 21 cases in the present study with an increased predilection for the left side (57.14%). With regard to the site of lesion in the jaw bones, maxillary and mandibular involvement noticed was 1.08:1. Among the cases involving the maxilla, the premolar–molar area was affected in five cases; the canine–premolar–molar area in three cases; the entire maxilla was affected in three cases; and the canine–premolar area alone was affected in one case. Among the cases affecting the mandible, the body of the mandible was involved in 12 cases; the angle region was affected in 4 cases; and the ramus in 3 cases.

Table 1. Summary of clinical and radiographic features of fibrous dysplasia affecting the mandible.

Case no. Age Sex Chief complaint with duration Site of the lesion Radiographic picture
Periphery Internal structure Effect on surrounding structure
1 28 Female Swelling 4–5 years 42–34 region (crossing the midline) Ill defined Ground glass appearance Displacement of 32, 31, 41, 42; loss of lamina dura of 33, 32, 31, 41, 42, 43.
2 20 Female Swelling 3 months 35–38 region Ill defined Mixed radiolucent–radiopaque (indiscernible pattern) Expansion of buccal cortex; inferior alveolar nerve canal displaced superiorly
3 55 Male Swelling 18 years 42–48 region Well defined Sclerosis Inferior alveolar nerve canal not discernible Loss of lamina dura on 43, 44, 45, 47
4 12 Female Swelling 6 months 43–48 region Ill defined Radiolucent Loss of lamina dura on 44, 45, 46
5 35 Male Swelling 6 years 33–37 region Ill defined Mixed radiolucent–radiopaque with cystic pattern Inferior alveolar canal displaced superiorly Displaced 35 Loss of lamina dura 33–37
6 15 Female Swelling 2 years 32 region to the left angle of the mandible Well defined Ground glass appearance Expansion of lower cortex of the mandible giving a characteristic “thumb-print” appearance. Loss of lamina dura on 35, 36, 37
7 39 Male Swelling 2 years 35–44 region (crossing the midline) Well defined Ground glass appearance Expansion of the labial cortex; inferior displacement of inferior alveolar canal. Loss of lamina dura of all associated teeth
8 64 Female Swelling and pain in left lower teeth 6 months 33 region to the left ramus of the mandible Ill defined Ground glass appearance Expansion of the buccal and lingual cortical plates with characteristic “thumb-print” appearance Inferior alveolar canal not discernible Superior displacement of 36, 37, 38 Loss of lamina dura of 33–38
9 32 Female Swelling 4 years 42–45 region Ill defined Mixed radiolucent–radiopaque (indiscernible pattern) Expansion of buccal cortical plate Displacement of 43, 44. Loss of lamina dura on 43, 44, 45
10 24 Female Swelling 6 years 46 region to the right angle of the mandible Ill defined Orange peel appearance Expansion of bucco-lingual cortical plates Expansion of lower border of posterior body and angle of the mandible on the right side Inferior alveolar canal superiorly displaced Loss of lamina dura on 46, 47
11 32 Male Swelling 5 years 48 region to posterior body of the mandible Ill defined Ground-glass appearance Inferior alveolar canal displaced inferiorly Loss of lamina dura of distal root of 47
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Table 2. Summary of clinical and radiographic features of cases affecting the maxilla.

Case no. Age Sex Chief complaint with duration Site of the lesion Radiographic picture
Periphery Internal structure Effect on surrounding structure
12 42 Female Swelling since childhood 14–18 region Ill defined Ground glass appearance Near total obliteration of right maxillary sinus Loss of lamina dura of 14
13 20 Female Swelling 5–6 years, associated pain in 27 region 4 days 22–27 region Ill defined Ground glass appearance Displacement of floor of the sinus Loss of lamina dura of 23–27
14 14 Male Swelling 4 years 25–28 region Ill defined Orange peel appearance Effacement of sinus walls; resorption of roots of 24, 25 Loss of lamina dura of 24, 25, 26
15 9 Male Swelling 6 months Right ala to zygoma region 11–16 region Ill defined Ground glass appearance Expansion of buccal cortex in relation to involved teeth Lamina dura loss on 12–16
16 10 Female Swelling 6 months 25–27 region Ill defined Orange peel appearance Loss of lamina dura of 26, 27
17 66 Male Swelling since second decade 22–28 region Ill defined Ground-glass appearance Sinus floor effaced Loss of lamina dura on 23–28 region
18 21 Female Swelling 1 month 25,26 region Ill defined Orange peel appearance Loss of lamina dura on 25, 26
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Figure 1.

Figure 1

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Axial CT scan of the skull in a case of craniofacial fibrous dysplasia. The involvement of the zygoma, the sinus and the anterior cranial fossa is seen

Table 3. Summary of jaw bone features in craniofacial and polyostotic lesions of fibrous dysplasia.

Case no. Age Sex Chief complaint with duration Site of the lesion Radiographic picture
Periphery Internal structure Effect on surrounding structures
19 13 Male Swelling with protusion of left eyeball 6 months Frontal bone, left maxillary sinus, left zygoma, roof of sphenoid Ill defined Sclerosis Effacement of sinus walls
20 21 Male Swelling with difficulty in mouth opening 6 months Left squamous temporal bone, left zygoma Ill defined Mixed radiolucent–radiopaque indiscernible pattern Expansion of the zygoma
21 54 Female Pain and swelling 6 months 14–25 region (crossing the midline) Frontal, zygomatic & nasal bone, anterior, middle posteror cranial fossa Ill defined Ground-glass pattern of skull bones Sclerosis frontal bones Cystic changes in the maxilla Displacement of 11 and 21 Loss of lamina dura of 14–25
22 21 Male Swelling 10 years 47, 48 region extending to the ramus, involving the posterior end of hard palate right side Ill defined Ground glass appearance Expansion of lower cortical plate of the mandible Inferior alveolar canal not discernible
23 35 Male Swelling 6 years 15–18 region, zygoma, frontal bone,temporal bone, maxillary sinus Ill defined Ground glass appearance Near total obliteration of the right maxillary sinus Expansion of buccal cortical plate of the right maxilla Loss of lamina dura of 16, 17, 18
24 32 Male Swelling left leg 6 months, swelling face 15 years 23–28 region 44–48 region to the sigmoid notch, ribs, femur Ill defined Mixed radiolucent–radiopaque indiscernible pattern Effacement of sinus walls Increased width of ramus and body Loss of lamina dura 24–27, 33–37
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Radiographic analysis of the lesion

Of all the cases reviewed, 12 cases were investigated by CT. Only two cases, both of which involved the mandible, showed a well-defined corticated border. The others had ill-defined peripheries. On studying the internal structure of the lesion on plain films, a single case depicted a purely radiolucent appearance. 19 cases showed a mixed radiolucent–radiopaque appearance, of which 4 cases showed a non-discernible pattern (Figure 2). The characteristic orange peel appearance (Figure 3) was noticed in 4 of the cases studied and a ground-glass appearance noticed in 11 cases. Sclerosis was noticed in three cases of fibrous dysplasia studied (Figures 4 and 5). Cystic change of the lesion was noticed in two cases, one each involving the mandible and the maxilla (Figure 6). Among the lesions involving the maxillary sinus, displaced borders of the sinus were noticed in five cases. Near total obliteration of the left maxillary sinus was noticed in two cases. Of the cases involving the mandible, expansion of the lower border of the mandible was noticed in four cases and the characteristic “thumbprint pattern” in two cases (Figure 7). In case 21, the CT revealed varied internal structure of bones; frontal bones showing dense sclerotic foci scattered in a ground-glass pattern and cystic changes around the root of maxillary teeth. In the cases involving the mandible, the inferior alveolar canal appeared to be superiorly displaced in three cases while it was inferiorly displaced in two cases (Figure 8). The canal was not traceable in three cases. On studying the teeth involved in the site of the lesion in these cases, root resorption was noticed in a single case involving the maxilla. Loss of lamina dura of the teeth was noticed characteristically in 20 cases.

Figure 2.

Figure 2

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Panoramic radiograph showing a mixed radiolucent–radiopaque appearance with an indiscernible pattern (arrow)

Figure 3.

Figure 3

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Intra oral periapical radiograph displaying an orange peel appearance. The inferior alveolar nerve canal (black arrows) has been displaced upwards markedly. Contrast has been enhanced to visualize the canal trajectory

Figure 4.

Figure 4

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Panoramic radiograph displaying the characteristic sclerosis. The borders of the lesion are well defined. The inferior alveolar nerve canal is obscured because of the lesion

Figure 5.

Figure 5

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Coronal CT of case 21. Frontal bones show heterogeneous round sclerotic foci scattered in ground glass pattern

Figure 6.

Figure 6

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Bone window axial CT scan of case 21 showing cystic changes in the lesion. The cystic changes appear to be confluent around the maxillary teeth

Figure 7.

Figure 7

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Part of a panoramic radiograph showing the “thumb print” pattern of the lesion in the mandible. Note that the inferior dental canal has been displaced inferiorly

Figure 8.

Figure 8

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Panoramic radiograph displaying the characteristic inferior displacement of the inferior alveolar nerve trajectory

Discussion

This clinical series demonstrates the analysis of the various presentations of fibrous dysplasia. There are variations in this study compared with the earlier reports and studies on fibrous dysplasia. The average age of presentation was 29.75 years. This was the age at which the cases presented to the clinic. When prior awareness of the patients was considered, the calculated mean age was found to be 19.2 years. Two patients over the age of 60 years presented with a massive swelling, indicating that such lesions may remain asymptomatic and may stop patients from seeking dental care until there is gross disfigurement or pain is experienced. In case 17, the patient gave a history of swelling of more than 40 years duration but refused treatment because the lesion was not increasing in size. This is in accordance with Eisenberg and Eisenbud, who stated that the majority of cases burn out in early adulthood when skeletal maturity has been reached.12 A single male patient in the fourth decade of life presented with the polyostotic form consistent with the findings of Windolz,13 who revealed that the polyostotic form typically represented only about one-third of patients. In our study, males were more affected, disparate with the systematic review of cases of fibrous dysplasia by MacDonald-Jankowski,14 Yoon et al15 and Slootweg and Muller,11 where a female predominance was noted. Unlike most studies on fibrous dysplasia, in our clinical series the mandible was more affected than the maxilla.11,14,15 A predilection for the left side was observed in this study; however, it may be expected that such a predilection may be just an earlier stage of manifestation of the disease condition in monostotic patients and may vary when the sample size is larger.14,16

Craniofacial bones were involved in 25% of the cases, including the case showing a polyostotic presentation (Table 3). With regard to involvement of the bones of the craniofacial region, the mandible was the most involved bone followed by the maxilla, zygoma, frontal, temporal, sphenoid, ethmoid, parietal and occipital bones. Such a presentation is in contrast with the findings of Lustig and co-workers,7 who identified the ethmoid as the most commonly involved bone, followed by the sphenoid, frontal, maxilla, temporal, parietal and occipital bones.

This article highlights the various radiographic presentations of fibrous dysplasia in the jaw bones. To a certain extent, plain films help to define the lesion. CT scans are required to demarcate lesions in the anatomically complex maxilla and the skull. Periapical radiographs however do help to determine the internal structure of fibrous dysplasia accurately as it is placed closest to the jaw. Only 2 of the 24 cases showed well-defined borders, both of which involved the mandible. The lesion may be sharply defined when a sclerotic rim is present or may be ill defined if it lacks perilesional sclerosis and may fade into the adjacent normal bone.17 Sherman and co-workers18 and Eversole et al19 reasoned that diffuse lesions are usually poorly defined and dysplastic in nature. The basic patterns described by Fries in 1957 are pagetoid (56%), a mixture of dense and radiolucent areas of fibrosis; sclerotic (23%), mass is homogeneously dense; and cystic (21%), a spherical or ovoid lucency surrounded by a dense boundary.20 In this clinical series, the most common presentation was that of a mixed radiolucent–radiopaque type pattern of which the “ground-glass” appearance was most common. In the study by MacDonald-Jankowski and Li21 and in the systematic review by MacDonald-Jankowski,14 fibrous dysplasia most commonly presented radiographically as a poorly defined ovoid (fusiform) area of dysplastic bone exhibiting a ground-glass appearance. Cystic change was noted in two cases in patients who were in their fourth and fifth decades of life, thus justifying that radiolucent lesion resembling cysts occasionally occur in mature lesions of fibrous dysplasia.22 In fibrous dysplasia of the mandible, only four cases showed expansion of lower border of the mandible of which the characteristic “thumb-print” pattern was noticed in two cases (Figure 7). Petrikowski et al23 suggested that the upward displacement of the inferior alveolar canal was a unique characteristic of fibrous dysplasia. In the present study, the inferior dental canal was superiorly displaced in three cases, inferiorly displaced in two cases and remained non-traceable in four cases, thereby agreeing with the findings of MacDonald-Jankowski and Li.21

Fibrous dysplasia affecting the maxilla always involves the antrum, either by obliterating it or by gross displacement of its borders encroaching on the neighbouring bones, as in the cases depicted in our study.9 Loss of lamina dura was noted in 20 cases, which has been considered as a pathognomonic sign on fibrous dysplasia.23

In conclusion, it may be emphasized that the radiographic study of these lesions, using both plain films and advanced methods like CT, contribute to the exact diagnosis and also aid in defining the accurate extent of the lesion. Aesthetic deformity or functional disturbances associated with the jaw lesions of fibrous dysplasia are the major indications for treatment. Awareness of the myriad of radiographic appearances of fibrous dysplasia is essential and will be beneficial in the accurate diagnosis and proper treatment planning, even without invasive diagnostic procedures.

Acknowledgments

We are grateful to Dr LS Sreela, Assistant Professor, Department of Oral Medicine and Radiology, Government Dental College, Kottayam; Dr Nileena R Kumar, Assistant Professor, Department of Oral Medicine and Radiology, Government Dental College, Kozhikode; Dr P Rani Mol, PG Student, Department of Oral Medicine and Radiology, Government Dental College, Trivandrum; and all others who allowed access to their file records of patients. We are also thankful to Dr VT Beena and Dr R Heera of the Department of Oral Pathology and Microbiology, Government Dental College, Trivandrum, for their support and co-operation.

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