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KDJ. Vol.7 No.1 January - June, 2004

Multidisciplinary Approach to the

Management of Fibrous Dysplasia of the

Maxilla : A Case ReportTanaporn Ruksujarit*, Somsak Kitsahawong**, Pornpaka Thongdee***

* Graduate student, Master of Science in Orthodontics, Department of Orthodontics, Faculty of Dentistry, Khon Kaen University

** Assistant Professor, Department of Orthodontics, Faculty of Dentistry, Khon Kaen University

*** Lecture, Department of Oral and Maxillofacial Surgery, Faculty of Dentistry, Khon Kaen University

Abstract

Fibrous dysplasia is a benign fibro-osseous lesion of bone that commonly affects the jaws with a

higher prevalence in the maxilla than the mandible. Based on a case of maxillary fibrous dysplasia in a Thai

patient, this article aims to elucidate the principles of diagnosis and treatment of fibrous dysplasia. Proper

integration of the clinical, radiographic, and histologic findings is essential for an accurate diagnosis of the

lesion. The dental surgeon who can be the health practitioner to first attend the patient should also be aware

that management of this disease entity may necessitate a multidisciplinary approach. Due to the difficulty

with prognosticating the recurrence of fibrous dysplasia, it is advisable to review the patient periodically for

any clinical and/or radiographic changes.

Key Words : fibrous dysplasia, maxilla, Thai, multidisciplinary management

Introduction

Fibrous dysplasia is a benign fibro-osseous lesion

of bone that is characterized by the replacement of bone

with abnormal fibrous connective tissue containing varying

amounts of calcification.1

It was first described as a disease

entity by Lichtenstein in 1938.2

The etiology of fibrous

dysplasia is unknown. Endocrine, infectious, traumatic and

neoplastic theories of origin had been suggested.3

One ofthe most widely accepted theory is that fibrous dysplasia is

caused by aberrant activity in the bone-forming mesenchy-

mal tissue, as indicated by Lichtenstein and Jaffe.4

Fibrous

dysplasia is divided into two major forms: monostotic and

polyostotic. Monostotic fibrous dysplasia, characterized by

involvement of only one bone, is considerably more

prevalent than the polyostotic type. The jaws are commonly

affected with the maxilla being more frequent than the

mandible. Monostotic fibrous dysplasia is more easily found

in children and young adults.1,4,5

Some authors report an

equal predilection for both sexes1,5,6

but others report a higher

incidence among females.3,7

Polyostotic fibrous dysplasia

is defined by having more than one skeletal involvement.

Bones of the face, skull, clavicle, and the long bones are

most often affected. A severe type of polyostotic fibrous

dysplasia, known as Albrights syndrome, is characterized

by the presence of skin lesions and precocious puberty in

girls. Skin lesions appear as light brown macules called

cafe au lait spots. Polyostotic fibrous dysplasia is more

commonly diagnosed in children and has a definite female

predilection.1,5

Clinically, a painless enlargement of the affected

bone is the most common presenting symptom. Bulging of

the canine fossa and hyper-prominence of the zygomatic

process of the maxilla are usual features of maxillary

lesions with frequent involvement of the maxillary sinus.

In the mandible, a swelling is most often found at the angleof the jaw.

3Fibrous dysplasia of the maxilla or mandible is

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usually unilateral.1

The lesion grows very slowly, causing

expansion of the involved bone and giving a non-tender

facial asymmetry of variable degree. As a rule of thumb,

the lesion usually stops growing when skeletal growth

ceases, but cases of continual enlargement have been

reported.4

The expansion is smooth and the overlying skin

or mucosa appears normal. Soft tissue ulceration over the

bony enlargement is uncommon but may be seen when the

mass disrupts the dental occlusion or is traumatized during

eating. Teeth in the area of involvement may show

minimal migration or displacement but are rarely loosened.

In children, teeth in the affected part may fail to erupt.

Laboratory assay of serum calcium and phosphate levels of

affected patients are normal, but the alkaline phosphatase

level may be slightly elevated. Conversely, lack of an

elevation should not rule out fibrous dysplasia.4

The radiographic appearance of fibrous dysplasia

is extremely variable and depends on the proportion and

distribution of fibrous tissue deposition. When the fibrous

tissue component predominates, the lesion may have a

cystic radiographic appearance, being either unilocular ormultilocular. Such radiographic features of fibrous dysplasia

are usually noted in younger patients or in those lesions of

shorter duration. Fibrous dysplasia having larger irregular

trabeculae assumes a mottled appearance. When the fibrous

elements have undergone considerable ossification, the

radiographic appearance is characterized by areas of con-

densation interspersing with areas of radiolucency, described

as ground-glass or orange-peel appearance.3,4

The more

osseous or highly calcified lesions of fibrous dysplasia are

usually found in older patients and in lesions of longer

duration. Among these three basic roentgenographic types

of fibrous dysplasia, the ground-glass appearance is the most

common. The cystic and the mottled lesions are usually

sharply delineated and surrounded by an osteosclerotic

margin. On the other hand, ground-glass lesions are not

well-circumscribed and blended almost imperceptibly into

the normal surrounding bone. Fibrous dysplasia may cause

root resorption of the erupted teeth on which it encroaches.3,4

Pathologically, fibrous dysplastic tissue has a

yellowish to grayish white macroscopic appearance, being

gritty on section.4

The classic histologic findings of fibrous

dysplasia in long bones include cellular fibrous connective

tissue often arranging in a whorled pattern and consisting of

irregularly shaped trabeculae of immature, woven bone. The

trabecular arrangement has been likened to the appearance

of Chinese script writing and is often referred to as

Chinese character trabeculae.5

Histologic features are

diagnostic for fibrous dysplasia of the jaws but are

considerably controversial for the skull. Some authors do

not accept the presence of lamellar bone and osteoblastic

rimming to be compatible with the diagnosis of fibrous

dysplasia.8,9

Others agree on a wider range of histologic

features.3,6

Fibrous dysplasia of the skull and jaw bones

tend to have a higher degree of ossification than their

counterparts in the long bones.5

Diagnosis is accomplished through evaluation of

the clinical, radiographic, laboratory and histologic findings.

Other lesions which may resemble fibrous dysplasia include

ossifying fibroma, cysts, cementomas, Pagets disease,cherubism, hyperparathyroidism, chronic sclerosing

osteomyelitis, osteogenic sarcoma and other fibro-osseous

lesions of the jaws.4,5,10,11

Treatment for fibrous dysplasia is almost always

surgical intervention. When the lesion is small and

well-delineated, curettage and enucleation or excision can

be accomplished without creating a large surgical defect or

loss of continuity of bone. If the lesion is extensive and

presents with malocclusion and jaw disproportion, conser-

vative recontouring surgery and repositioning of the affected

jaw bone aiming at aesthetic correction or functional

improvement are recommended.4,7,12

In most instances,

fibrous dysplastic tissues from affected jaws are clinically

softer and less vascular than normal bone and present with

minimal bleeding during recontouring.13

In children, it is

recommended to delay the surgical procedure until after

puberty, so that the lesion tends to become static.

Occasionally, the deformity may be so cosmetically

unacceptable that earlier intervention is indicated.3,4

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According to Zimmerman et al3, approximately 20% of the

lesions will continue to grow after treatment, unless radical

excision is performed. Radiotherapy in the treatment of

fibrous dysplasia is contraindicated because of the possibility

of radiation induced sarcomas.14

Because of the lack of

means to prognosticate the recurrence of fibrous dysplasia,

all patients should be periodically evaluated both clinically

and radiographically.1,4

Based on a case report and discussion, this article

aims to elucidate the principles of diagnosis and treatment

of fibrous dysplasia and demonstrate the importance of a

multidisciplinary approach to the management of this

disease entity.

Case Report

Case history

A 19 years old Thai female was referred to the

Orthodontic Clinic, Faculty of Dentistry, Khon Kaen

University in May 2002 for her chief complaint of facial

asymmetry from a bony swelling on the right side of the

upper jaw and the lingual crossbite of teeth #12 to 15. Ingeneral, patient was healthy but her medical history was

highlighted by two previous operations in the concerned

orofacial region. Accordingly, surgical recontouring was

done transorally when she was 11 years old and again via

the Weber-Ferguson extraoral approach at the age of 17

years. Patients parents recalled that the lesion was first

being aware when their daughter was 8 years old. Since

that time, the lesion had been slowly enlarging and became

relatively static after the second surgery. There was no

history of dentofacial trauma or previous orthodontic

treatment. Tooth #45 had been extracted a few months ago

because of malposition and lack of space for eruption. Her

family history was unremarkable.

Clinical evaluation

Extra-oral assessment

A non-tender bony enlargement was found on the

right face extending horizontally from the posterior malar

to the nasal region and vertically from the infraorbital

region to the maxillary alveolus, causing eversion of the

upper lip (Fig. 1A). There was a 10 mm difference in antero-

posterior dimension between the affected and non-affected

side of the face (Fig. 1B). The patient presented asymmetric

tapered facial form at the midfacial and upper lip areas with

increased lower anterior facial height. The lips were

incompetent with 7 mm incisal show at rest (Fig. 1A) and 6

mm above the gingival margin on smiling, revealing a

gummy smile (Fig. 1C). The chin position coincided with

the facial midline (Fig. 1A). The upper dental midline was

deviated 1 mm to the left and the lower one to the right by 5 mm

(Fig. 1C). For the lateral profile, the patient had a normal

convexity with flat dorsum of nose, turn-up nasal tip,

normal nasolabial angle, everted upper lip, protruded upper

and lower lips, slightly deep mentolabial fold, retrusive chin

position and steep mandibular plane angle (Fig. 1D and 1E).

Her maximum mouth opening was normal at 46 mm. There

was no sign and symptom of the temporomandibular joints.

Her speech was normal. There was both nasal and oral

breathing.

Intra-oral assessment

There was buccal cortical expansion on the rightmaxillary alveolus in the region of tooth #12 to tooth #16.

Scarring from previous surgery was found on the covering

mucosa but without any tenderness to palpation. The

patient had a good oral hygiene and healthy gingival status.

Clinically, tooth #45 and the third molars were missing.

Teeth #12 to 15 exhibited crossbite relationship with their

occluding lower teeth. The maxillary dental arch was

asymmetric and tapered and the mandibular arch ovoid. The

upper lateral incisors were peg-shaped and tooth #15 was

slightly rotated. There was mild crowding (1 mm) in the

anterior mandibular arch. All lower premolars were rotated,

except tooth #45 which as aforementioned, had been

extracted. There was Class I molar and Class II canine

relationship on the right side and Class III molar and canine

relationship on the left side. The overbite was 3 mm (30%

of lower incisal crown) and so was the overjet. The curve of

Spee was 2 mm on each side. There was a discrepancy of 2

mm between centric relation (CR) and centric occlusion (CO)

(Fig. 2).

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A B C

D E

Fig. 1. Pre-treatment extraoral facial photographs. (A) Frontal view in resting position. (B) Supero-inferior view. (C) Frontal view in smiling

position. (D) Profile view on the affected (right) side. (E) Profile view on the normal (left) side

A

B C

D E

Fig. 2. Pre-treatment intraoral photographs. (A) Frontal view. (B) Lateral view-right side. (C) Lateral view-left side. (D) Occlusal view-

upper arch. (E) Occlusal view-lower arch.

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Radiographic evaluation

Panoramic radiograph

The orthopantomogram revealed a ground-glass

appearance of the right maxilla, extending from the lateral

incisor to the third molar region. The right maxillary sinus

and part of the nasal cavity appeared obliterated. The

ill-defined radiopacity merged imperceptibly with the

surrounding bone. Teeth #18, 28 and 38 were unerupted

and impacted, and tooth #48 was congenitally missing. All

erupted teeth demonstrated normal crown-root ratio and were

supported in normal bone levels. The mandibular condyles

appeared normal (Fig. 3).

Lateral cephalogram

The lateral cephalometric measurements are

shown in Table 1. Analysis of the measurements indicated

skeletal Class I relationship with increased lower anterior

facial height and steep mandibular plane angle. The maxillary

and mandibular incisors were normal in both inclination and

position. The facial profile showed a normal convexity,

normal nasolabial angle, protruded upper and lower lips and

retrusive chin position (Fig. 4).

Fig. 3. Pre-treatment panoramic radiograph.

Postero-anterior or frontal cephalogram

Assessment of frontal cephalogram was based on

Grummons analysis. The frontal cephalometric radiograph

indicated asymmetry in the middle and lower facial thirds

and mild maxillary canting with compensatory mandibular

canting. The chin point (menton) was deviated 4 mm to the

right. The upper dental midline was deviated 1 mm to the

left, while the lower one was deviated 6 mm to the right

(Fig. 5).

Computed tomography (CT) scan

CT scan revealed skeletal deformity of the right

face. Features of expansion, sclerosis with ground glassappearance and bone destruction with widened diploic space

were noted in the right maxilla (palatine and frontal

processes and the superior alveolar arch), the ethmoid bone,

the palatine bone, the sphenoid bone (pterygoid plate and

the lesser and greater wings) and the zygomatic bone.

Narrowing of the right orbit and orbital apex was observed.

Figure 6 illustrated the obliterated right maxillary sinus and

sphenoid air cells.

A B

Fig. 4. Pre-treatment lateral cephalometric radiograph (A) and tracing (B).

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Measurements Thai norm Pre-treatment 7-week post-surgery

Skeletal:

SN length (mm) 70 4 64.5 64

SN-FH () 5 6* 5 5.5

SNA () 85.4 4 86 85

SNB () 81 3.7 82 80

ANB () 3.8 2 4 5

SN-MP () 29 4 35 37.5

UAFH:LAFH (%) 45:55* 42:58 42:58

PFH:AFH (%) 67 5 63.0 60.9

Gonial angle () 120 6 123.5 122.5

Dental:

U1-SN () 107 6 102 103.5U1-NA () 21 2 15 18

U1-NA (mm) 3 2 4 4

L1-MP () 97 6 96 97

L1-NB () 30 5 34 36

L1-NB (mm) 6 2 8 9

Soft tissue:

Profile angle () 168.7 4* 167 165

Nasolabial angle () 102 8* 93 103

U-lip to E-line (mm) -1 2 3.5 3

L-lip to E-line (mm) 1.5 2 5.5 6

Pog' to SnV (mm) -1-(-4)* -10 -10.5

U-lip length (mm) 20.1 1.9* 22 24

L-lip length (mm) 46.4 3.4* 48 47.5

* represent Caucasian norm.

Table 1. Lateral cephalometric measurements of the patient.

A B

Fig. 5. Pre-treatment frontal cephalometric radiograph (A) and tracing (B).

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Hand-wrist radiograph

The hand-wrist radiograph indicated fusion of the

radius (Fig. 7), corresponding to skeletal maturity

indicators (SMI) 11 of Fishmans skeletal maturity indicators.

This finding was compatible with completed skeletal growth.

A B

Fig. 6. Pre-treatment CT scan. (A) Axial view. (B) Coronal view.

Histological examination

The biopsy report documented irregularly shaped

trabeculae of immature bone in a cellular loosely arranged,

fibrous stroma. The bone trabeculae were not surrounded

by osteoid seams or appositional osteoblasts. Reversal lines

were evident in most of the trabeculae and scattered

erythrocytes could also be seen (Fig. 8). In conjunction with

the age of the patient and the clinical and radiographic

findings, a histopathologic diagnosis of fibrous dysplasia

was reached.

Treatment

The following treatment objectives were defined:

1. To correct facial asymmetry.

2. To correct maxillary lingual crossbites of teeth

#12 to 15.

3. To align and level all teeth.

4. To obtain optimal overbite and overjet.

5. To obtain Class I molar and canine relationship

on the right side and accept a compromised

occlusion on the left side.

6. To obtain a normal maxillary and mandibular

anterior tooth proportion by building up the

maxillary lateral incisors with composite

filling

During consultation, the patient and her parents

were informed that the facial asymmetry and maxillary

lingual crossbites required orthodontic correction with

adjunctive orthognathic surgery. Treatment was started with

pre-surgical orthodontics using fixed appliances to align and

Fig. 7. Pre-treatment hand-wrist radiograph.

Model analysis

The maxillary dental arch had no space problem

but the mandibular anterior teeth had mild crowding of

1 mm. Boltons anterior ratio analysis indicated an excess

of 4.3 mm in the size of mandibular anterior teeth (anterior

ratio 87.2%). The great discrepancy of Boltons anterior

ratio came from the small size of the peg-shaped upper

lateral incisors. In order to achieve an incisal Class I rela-

tionship and proper occlusion in the buccal segments, the

width of the maxillary lateral incisors had to be built up.

A joint consultation with the oral and maxillofacial

surgery specialty was arranged and an incisional

biopsy was performed.

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level teeth. This was followed by orthognathic surgery

involving firstly a maxillary segmental osteotomy in the area

of 12-15 to correct the crossbite and secondly, surgical

recontouring of the enlarged right maxillary buccal alveolar

process (extending from the incisal region to the

tuberosity) and zygomatic process.

Pre-surgical orthodontic phase

The patient had a fixed appliance 0.022' x 0.028'

Roth prescription placed in June 2002. The orthodontic treat-

ment commenced with aligning all teeth in the lower arch

by 0.014' NiTi archwire. The patient was asked to wear a

lower posterior raising-bite appliance to reduce the occlusal

interference at the crossbite area while aligning the upper

arch with 0.014' NiTi. Sequentially, the upper and lower

archwires were changed to stainless steel wires of 0.016',

0.016' x 0.022' and 0.019' x 0.025' respectively. Two

weeks before surgery, surgical hooks were placed on the

archwires and the upper archwire was segmented between

tooth #11 and #12 and between tooth #15 and #16 to facili-tate the operation. Model surgery was also done to evaluate

and establish the proper occlusion.

Surgical phase

The orthognathic surgery was performed in

October 2003 and the procedures were accomplished as

planned. Rigid fixation was achieved by using miniplate

and screws without intermaxillary fixation or placement of

surgical splint. Teeth #18, 28 and 38 were also extracted in

the operation.

Post-surgical orthodontic phase

The post-surgical orthodontic treatment began

after the osteomized dentoalveolar segments were stabilized

for seven weeks. Because the mandibular dental midline

shift (5 mm to right side) was almost equal to the width of

one lower incisor, the lower dental midline at the comple-

tion of orthodontic treatment was designed to be between

tooth #31 and #32. Consequently, teeth #42 and #34 would

be positioned as resembling the mandibular canines. The

maxillary lateral incisors had to be built up after finishing

orthodontic treatment to obtain a normal maxillary and

mandibular anterior tooth proportion. Wrap-around retain-

ers were to be used for retention in the upper and lower

dental arches. Periodic follow-up at every six months was

planned in order to monitor the stability of the occlusion

and any further growth of the lesion.

Results

This article will only present the treatment

outcome after surgery but not at the finishing stage because

the post-surgical orthodontic treatment is still in progress.

At seven weeks after operation, clinical and radiographic

assessment confirmed achievement of the treatment objec-

tives. Clinically, the patient was satisfied with her state of

facial symmetry even there was mild residual prominence

on the right side of her face and mild improvement in her lip

competence (Fig. 9A). Supero-inferior view of the face

demonstrated comparable contour of the right and left

zygomatic areas (Fig. 9B). Although she still presented

Fig. 8. Histologic view of the biopsy. Hematoxylin-eosin stain. Original magnification, (A) x 10 and (B) x 40.

A B

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a gummy smile, her dental occlusion and midlines were

much better on smiling (Fig. 9C). On lateral view, improve-

ment in the right malar region was evident though the other

parts of her face were similar as pre-treatment (Fig. 9D and

9E). Intraorally, the maxillary lingual crossbite was

corrected. The patient had normal occlusion with symmetric

maxillary and mandibular arches, overbite of 2 mm, overjet

of 3 mm and absence of crowding in the lower arch

(Fig. 10).

Lateral cephalometric analysis indicated significant

change in dental and soft tissue relationship after pre-surgical

orthodontic treatment and surgery. The upper and lower

incisors became more proclined and the upper first molars

were moved forward by approximately 2 mm. Her upper

lip was more competent and retrusive, resulting in an

increased nasolabial angle (Fig. 11 and Table 1). The

postero-anterior cephalometric radiograph taken at 7 weeks

after surgery revealed more symmetric lower facial third

and less deviated chin point (3 mm to the right). The upper

dental midline was slightly deviated 0.5 mm to the left while

the lower one became less deviated to the right (5 mm).

Her overbite was also reduced (Fig. 12). In the time being,

the patient has started post-surgical orthodontic treatment

with stainless steel continuous wire of 0.016" in the upper

arch and 0.016" x 0.022" in the lower arch.

Fig. 9. Extraoral facial photographs at 7-week post-surgery. (A) Frontal view in resting position. (B) Supero-inferior view. (C) Frontal view

in smiling position. (D) Profile view on the affected (right) side. (E) Profile view on the normal (left) side.

A B C

D E

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Fig. 10. Intraoral photographs at 7-week post-surgery. (A) Frontal view. (B) Lateral view-right side. (C) Lateral view-left side. (D) Occlusal

view-upper arch. (E) Occlusal view-lower arch.

A

B C

D E

Fig. 11. Lateral cephalometric superimposition between pre-treatment and 7-week post-surgery.

Pre-treatment -- -- -- -- -- 7-week post-surgery

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A B

Fig. 12. Frontal cephalometric radiograph at 7-week post-surgery (A) and tracing (B).

Discussion

According to Firat and Stutzmancited in

15

, fibrous

dysplasia accounted for 2.4% of all tumorous and tumor-

like bone lesions. Phaonimmongkol et al16

found only 70

Thai patients diagnosed as fibrous dysplasia from 1965 to

1995 in the Department of Oral Pathology, Faculty of

Dentistry, Chulalongkorn University. Even though the

incidence of fibrous dysplasia is low, a dental surgeon should

have the knowledge and competence to diagnose this

disease relating to the jaws. Historical, clinical, radiographic

and histologic data are all essential to confirm the diagnosis

of fibrous dysplasia. The patient reported in this article had

the lesion slowly enlarging on the right side of the upper

jaw without causing any discomfort since she was young.

The panoramic radiograph revealed a ground glass

appearance of the affected area. The histopathologic findings

documented irregularly shaped trabeculae of immature bone

in a fibrous stroma. All these were compatible with a diag-

nosis of monostotic fibrous dysplasia.

In the majority of patients with fibrous dysplasia,

surgical reduction for cosmetic or functional reason was the

treatment of choice. It is usually recommended to delay the

surgical procedure until skeletal growth has completed, so

that the lesions tended to be more static. Assessment of the

disease activity or stability can be based on subjective

evaluation of the patient with regard to the patients age,hand-wrist radiography and any alteration in size of the

lesion. For the present case, the result after evaluation of the

patient indicated static growth of the lesion. Consequently,

the third surgical intervention was decided to implement.

Zimmerman et al3

found approximately 20% of the lesions

would continue to grow after treatment, unless radical

excision was performed. Nevertheless, surgical excision was

only suitable for non-extensive and/or well-delineated

lesions. Because of the limitation of treatment and lack of

means to prognosticate the recurrence of fibrous dysplasia,

all patients should be periodically evaluated both clinically

and radiographically. Thus, the patient reported in this

article is expected to be followed-up in the long-term even

after finishing the prescribed treatment.

It has to be admitted that treatment selected for

this patient was a little compromised, as not all presenting

deformities were corrected (such as her gummy smile and

mild canting of the jaws and occlusal plane). In fact, patient

had emphasized her intention of avoiding more complicated

surgery during orthodontic-orthognathic joint consultation.

She readily accepted the proposed treatment plan which

focused on correcting her dentofacial asymmetry and

maxillary lingual crossbite. She was also satisfied with the

treatment outcome up to the current stage. Therefore, the

most appropriate treatment plan for a patient is not always

and merely the ideal plan but one which always meets the

chief complaint or concern of the individual.

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The success of treatment comes from an accurate

diagnosis, well executed treatment plan and collaboration

among related expertise such as orthodontist, oral and

maxillofacial surgeon, plastic surgeon, prosthodontist and

general dental practitioner. Care for the patient in this

article has been under the joint effort of an orthodontist, an

oral and maxillofacial surgeon and a restorative dentist.

Orthodontic treatment helped to facilitate the operation in

achieving the desired results and an optimal dental occlusion

after surgery, thus reaching the objectives of treatment.

Surgery was the main intervention to correct other dentofacial

deformities that could not be done by orthodontic

treatment alone. The restorative dentist helped to improve

the dental esthetic outcome in the final stage. Therefore,

the multidisciplinary team is crucial for optimal manage-

ment of fibrous dysplasia patients.

Conclusion

A case of fibrous dysplasia involving the maxilla

in a Thai patient was presented. Because of the tendency of

this disorder to affect the jaws, the dental surgeon must be

able to integrate the clinical, radiographic, and histological

findings, thus enabling an accurate diagnosis. Appropriate

treatment for this case involved orthodontic treatment,

maxillary segmental osteotomy in the dentoalveolar area of

#12 to 15, recontouring of the enlarged right maxillary

buccal alveolar process and restorative bui lt-up of

peg-shaped lateral incisors . All these imply a

multidisciplinary approach. The possibility of disease

recurrence mandates long-term patient follow-up.

Acknowledgements

The authors would like to express sincere gratitude

to Dr.Albert C.F. Leung for his valuable suggestion and

advice and Dr.Ajiravudh Subarnbhesaj for his useful

histological document.

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Correspondence author :

Assistant Professor Somsak Kitsahawong

Department of Orthodontics,

Faculty of Dentistry,Khon Kaen University

Khon Kaen, 40002, Thailand

Tel: (66) 4334-8309Fax: (66) 4324-4465

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