Vol.2, No.8, 484-489 (2013) Case Reports in Clinical Medicine
http://dx.doi.org/10.4236/crcm.2013.28127
The sequential treatment of extensive mandibular
ameloblastoma
Ruixiu Cheng1, Xingmao Fu2, Jianhui Ma1, Shuliang Li2, Jie Yu1, Kun Yan1, Yang Xue3*
1Department of Stomatology, The 89th Hospital of PLA, Weifang, China
2Institute of Traumatic Orthopedics, The 89th Hospital of PLA, Weifang, China
3Department of Oral Biology, School of Stomatology, The Fourth Military Medical University, Xi’an, China;
*Corresponding Author: xueyangfmmu@live.cn
Received 24 September 2013; revised 24 October 2013; accepted 5 November 2013
Copyright © 2013 Ruixiu Cheng et al. This is an open access article distributed under the Creative Commons Attribution License,
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
ABSTRACT
Ameloblastoma is the most frequent odonto-
genic tumor of the jaw. If not treated, amelo-
blastoma can gain an enormous size and cause
severe facial disfigurement and functional im-
pairment. Here we report two patients afflicted
with extensive mandibular ameloblastoma (sized
in 23 cm × 18 cm × 17 cm and 15 cm × 12 cm ×
10 cm, respectively). Both the patients received
the same sequential treatment including radical
tumor resection, simultaneous reconstruction
with fibula free flap graft, vertical distraction
osteogenesis on the fibula graft, placement of
endosseous dental implants, and final pros-
tho dontic rehabilitation. It took about 15 months
to finish the entire cours e of treatme nt. And af te r
the four-year follow-up, neither soft tissue re-
lated, nor hard tissue related problems were
observed. Satisfactory facial symmetry, chewing
and speech functions of the patients were re-
stored. So this sequential treatment for exten-
sive mandibular ameloblastoma can obtain an
excellent effect by the shortest time and the
lowest economical cost. Furthermore, the series
also can be used to reconstruct giant mandibu-
lar defects caused by different reasons.
Keywords: Ameloblastoma; Mandibular Defect;
Fibula Flap; Sequential Treatment
1. INTRODUCTION
Ameloblastoma, a benign but locally aggressive tumor
of odontogenic epithelium, is the most frequent odonto-
genic tumor of the mandible and maxilla [1]. It accounts
for about 1% of all tumors and cysts of the jaw and about
10% of the odontogenic tumors. Ameloblastoma is
mainly encountered during the third to the fifth decade of
life, with equal sex predilection [2,3].
According to different clinical and pathological mani-
festations, ameloblastomas are typically classified into 3
categories: multicystic/solid, unicystic and peripheral/
extraosseous ameloblastomas [2,4]. Multicystic amelo-
blastoma is the most common variety with an incidence
between 63.1% and 85% [4-6]. Unicystic ameloblastoma
is a less encountered variant with an incidence between
5% and 15%. It refers to those cystic lesions that show cli-
nical and radiographic characteristics of an odontogenic
cyst but histopathologically show a typical ameloblas-
tomatous epithelium lining part of the cyst cavity, with or
without luminal and/or mural tumor proliferation [7,8].
Peripheral ameloblastoma presents 1.3% to 10% of the
cases, showing histological characteristics of intraosse-
ous ameloblastoma that occur solely in the soft tissues
covering the tooth-bearing parts of the jaws [2].
The treatment of ameloblastoma mainly relies on sur-
gical operation. However, different operations depend on
different tumor types. Peripheral ameloblastoma can be
treated with conservative approaches such as enucleation,
while unicystic ameloblastoma can be treated with cu-
rettage [9]. However, multicystic ameloblastoma requires
radical extensive excision. Conservative treatments for
this type often lead to recurrence rates between 75% and
90% [4-6]. If not treated, the tumor can gain an enor-
mous size and cause severe facial disfigurement and
functional impairment.
The aim of the present report is to show the results of
a sequential treatment of patients with huge multicystic
ameloblastoma. The sequential treatment includes radical
tumor resection, simultaneous reconstruction with fibula
free flap graft, distraction osteogenesis (DO), placement
of endosseous dental implants and final prosthodontic
rehabilitation.
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R. X. Cheng et al. / Case Reports in Clinical Medicine 2 (2013) 484-489 485
2. CASE PRESENTATION
2.1. Case 1
A 46-year-old male patient presented to our depart-
ment with an about 23 cm × 18 cm × 17 cm mass in the
body of mandible (Table 1, Figures 1(A)-(C)). Lateral
cephalograms and computerized tomographic (CT) scan-
ning with 3 dimension reconstruction demonstrated a
great radiolucent region in the mandibular area including
the body and ramus (Figures 2(A) and (B)). After a se-
quence of treatments, including radical tumor resection,
simultaneous reconstruction with fibula free flap graft,
DO and dental implantation, satisfied appearance and
mandible function were achieved (Figures 1(D)-(I)).
At the end of the 4-year follow-up, there was no evi-
dence of tumor recurrence and integration of grafted tis-
sue, steady levels of bone around the fixtures, healthy
peri-implant tissues and satisfied occlusioon were found
(Figure 3).
Figure 1. Clinical manifestations of Case 1. (A)-(C): Preopera-
tively, the patient showed an about 23 cm × 18 cm × 17 cm
mass in the body of mandible in anterior view or lateral view.
(D)-(F) Two weeks after tumor resection and simultaneous
reconstruction with fibula flap, the patient’s appearance was
well restored; (G)-(I) The patient’s appearance after final
prosthodontic rehabilitation.
Figure 2. Radiological findings of Case 1. Preoperative CT
scanning with 3 dimension reconstruction (A) and lateral
cephalogram (B) demonstrated a great radiolucent region in the
mandibular area including the body and ramus. CT angiogra-
phy (C) at 2 weeks after tumor resection and reconstruction
with fibula flap showed smooth flow in transplanted peroneal
artery and direct plate fixation between the lower ends of the
osteotomy in right ramus and fibula bone cortex. The pano-
ramic radiograph (D) at 2 weeks after fibula reconstruction
showed obvious sutures at osteotomy sites. Six months after
fibula reconstruction, the panoramic radiograph (E) showed
good bone healing at osteotomy sites unless the site between
the lower ends of right ramus and fibula bone cortex. After
three-month consolidation, the panoramic radiograph (F)
showed good osteogenesis in the distraction gap. The last
panoramic radiograph (G) showed placement of dental implants
in the distracted fibula graft.
Table 1. The general information of the two patients.
Case 1 Case 2
Gender male Male
Age (year) 46 47
Tumor growth time (year) 18 3
Rapid growth time (year) 2 1
Tumor volume (cm3) 23 × 18 × 17 15 × 12 × 10
Tumor weight (kg) 2.7 0.9
2.2. Case 2
A 47-year-old male patient presented to our depart-
ment with facial asymmetry and an about15 cm × 12 cm
× 10 cm mass mainly in the left side of mandible (Table
1, Figures 4(A)-(C)). CT scanning with 3 dimension
reconstruction showed a great multicystic bulging mass
mainly in the left mandibular area including the body and
ramus (Figure 5(A)-(D)). After the same sequence of
treatments, satisfied appearance and mandible function
were achieved (Figure 4(D)-(I)).
Copyright © 2013 SciRes. OPEN ACCESS
R. X. Cheng et al. / Case Reports in Clinical Medicine 2 (2013) 48 4-489
486
Figure 3. Clinical and radiological findings of Case 1 at the
end of the 4-year follow-up. (A)-(D) Appearance of the patient
at the end of the 4-year follow-up. (E)-(G) Satisfactory occlu-
sion was achieved after placement of implants and prosthetic
restoration. H: The intraoral photo showed the soft tissue con-
ditions. (I)-(K) The radiological findings showed integration of
grafted tissue, steady levels of bone around the fixtures and
osseointegration of dental implants.
3. THE SEQUENTIAL TREATMENT
A treatment panel composed of oral and maxillofacial
surgeons, orthopedists, anaesthetists and dentists were
established preoperatively. And the same sequential treat-
ment (Table 2), including radical tumor resection, si-
multaneous reconstruction with fibula free flap graft,
distractor implantation, DO, distractor removal, place-
ment of endosseous dental implants and prosthodontic
rehabilitation, was applied to both the two patients.
3.1. Step 1 Tumor Resection
Under general anesthesia, gross total removal of the
tumor was performed by oral and maxillofacial surgeons.
In both the two patients, the condylars had not been in-
vaded by the tumor, so the condylars did not be removed
in the tumor resection (Figures 2 and 5).
3.2. Step 2 Reconstruction with Fibula Free
Flap Graft
Preoperatively, mandible plaster model was made ac-
cording to the proportion of the upper and lower jaw and
CT findings. The length of the fibula flap and osteotomy
position was determined according to the lower edge of
the plaster model. Orthopedists were responsible for cut-
ting the fibula flap, while the oral and maxillofacial sur-
geons participated in its shaping (Figure 6( A) ). In order
to reconstruction the height of mandibular ramus, direct
plate fixation was made between the lower ends of the
osteotomy in ramus and fibula bone cortex (Figure 2(C)).
Then microsurgical vascular anastomosis was made. The
oral mucosa and the skin incision were primarily closed.
Postoperative course was uncomplicated. CT angiogra-
phy at 2 weeks after operation showed smooth flow in
transplanted peroneal artery (Figure 2(C)).
Figure 4. Clinical manifestations of Case 2. (A)-(C) Preopera-
tively, the patient showed an about 15 cm × 12 cm × 10 cm
mass in the left side of mandible in anterior view or lateral view.
(D)-(F): Six months after tumor resection and reconstruction
with fibula flap, the patient’s appearance was well restored.
(G)-(I) The patient’s appearance after final prosthodontic reha-
bilitation.
Table 2. The sequential treatment list.
Treatment Interval between treatments
Step 1Tumor resection
Step 2Reconstruction with fibula
free flap graft Simultaneous with step 1
Step 3Distractor implantation 6 Months after step 2
Step 4Distraction osteogenesis 1 Week after step 3
Step 5Distractor removal 3 Months after step 4
Step 6Placement of endosseous
dental implants 3 Months after step 5
Step 7Prosthodontic rehabilitation 3 Months after step 6
3.3. Step 3 Distractor Implantation
Six months later, the patients received distractor im-
plantation under general anesthesia. When the mandible
was exposed, optimal integration of grafted tissue and
Copyright © 2013 SciRes. OPEN ACCESS
R. X. Cheng et al. / Case Reports in Clinical Medicine 2 (2013) 484-489 487
Figure 5. Radiological findings of Case 2. Preoperative CT
scanning with 3 dimension reconstruction (A)-(D) demon-
strated a great multicystic region mainly in the left mandibular
area including the body and ramus. The panoramic radiograph
(E) showed good bone healing and placement of dental im-
plants in the distracted fibula graft.
Figure 6. Intra-operative photographs of Case 2. A showed the
cutting and shaping of the fibula. B showed good contour and
integration of grafted fibula before placement of distractor. C
showed inverted trapezoidal osteotomy in the front jaw and 2
implanted distracters.
the steady levels of bone around the fixtures could be-
seen (Figure 6(B)). In order to prevent interference with
each other during the distraction, inverted trapezoidal
osteotomy was made in the front jaw, and then 2 distrac-
tors were implanted (Figure 6(C)). Rod of the distractors
pointed to the function surface of maxillary teeth.
3.4. Step 4 Distraction Osteogenesis
After a latency period of 7 days, the distractor was ac-
tivated 3 times daily (1.0 mm per day). The duration of
distraction lasted for 10 - 15 days until the height of front
jaw reaching 2.5 - 3 cm.
3.5. Step 5 Distractor Removal
After a consolidation period of 3 months, the patient
underwent the fifth surgical stage, removal of the dis-
tractor through the submandibular incision. New bone
with sufficient volume and density, which was depicted
radiographically was confirmed clinically during the
process of removing distractors.
3.6. Step 6 Placement of Endosseous Dental
Implants
Dental rehabilitation plan was conducted 3 months af-
ter the distractor removal. Following clinical and radio-
logical evaluations, the patients underwent vestibular
plasty and placement of endosseous dental implants. Two
and four OSSTEM GS implants with a diameter of
4.5 mm and a length of 11.5mm (Manufacturer OSSTEM
IMPLANT Co., Ltd.#507-8, Geoje 3-dong, Yeonje-gu,
Busan, Korea) were placed into the reconstructed area
for Case 1 and 2, respectively, according to the manu-
facturer’s instructions. Locations of the implants were
determined by temporary denture, while long axis of the
implants point to the function surface of maxillary teeth.
3.7. Step 7 Prosthodontic Rehabilitation
The final prosthetic rehabilitation was performed 3
months after the placement of implant. Ball attachment
was chose for further prosthodontic rehabilitation.
At the end of the sequential treatment, chewing and
speech functions of the patients were restored, without
surgical complications. The esthetic and functional out-
comes restituted a satisfactory quality of life to the pa-
tients. The four-year follow-up proved the optimal inte-
gration of grafted tissue and the steady levels of bone
around the fixtures. Peri-implant soft tissues are healthy.
4. DISCUSSION
Ameloblastoma is the most common tumor of odon-
togenic origin. Clinically, it is a painless, slow-growing
and persistent lesion, but behaves as an invasive and re-
curring tumor in spite of its benign histological nature
[1,2,10]. If not treated, ameloblastoma can gain an
enormous size and cause severe facial disfigurement and
functional impairment [9]. Both the patients reported
here delayed treatment because of economic reasons; as a
result, the tumor grew to such an extent and impacted
their life seriously.
Recurrence is another terrible feature of ameloblas-
toma. It has been reported that more than 50% of recur-
rence appears within the first 5 years after primary sur-
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R. X. Cheng et al. / Case Reports in Clinical Medicine 2 (2013) 48 4-489
488
gery [2], and the risk of recurrence mainly depends on
the method of treatment and the type and size of the tu-
mor [1,10]. From a surgical standpoint of view, it has
been reported that the recurrence rate following a simple
curettage is much higher than radical resection, 75% and
20%, respectively [1]. So radical resection was applied to
both the two patients reported in this article. And after a
4-year follow-up, no evidence of tumor recurrence was
found in the patients. However, radical surgical ablation
procedures will inevitably result in large tissue defects,
which lead to severe aesthetic and functional sequelae,
with a significant loss in the quality of life unless it is
reconstructed successfully [9]. The patient’s postopera-
tive quality of life largely depends on the quality of the
mandibular reconstruction. So how to restore the pa-
tient's appearance and mandible function is the key to the
treatment of huge ameloblastoma.
In recent years, vascularized bone grafts are widely
considered as a reliable technique for reconstructing
segmental mandible defects. Because in comparison to
non-vascularized free bone grafts, microsurgical transfers
of free bone grafts can reconstruct mandible defects with
an immediate source of blood supply to the graft [2].
Fibula, ilium, scapula, and radius are the 4 commonly
used osteocutaneous flaps for mandibular reconstruction
[9]. Among these flaps, fibula and iliac crest are the most
commonly used free flaps. In this series, we chose fibula
flap rather than iliac crest, giving full consideration to
fibula’s unique advantages, including providing suffi-
cient bone segment for any length of mandibular defect,
possibility of multiple osteotomies because of both en-
dosteal and segmental blood supply, re-establishing the
contour of the mandible, correction of intermaxillary
relation, less resorption, suitable for insertion of dental
implants because of its proper bone thickness and
bi-cortical structure, flap dissection under tourniquet
with minimal blood loss, and a long pedicle up to 8 cm in
length [2,9]. Furthermore, the skin flap of the fibula is
thin and pliable which is suitable for use as oral lining
[9]. And the four-year follow-up of the patients proved
that continuity and contour of the mandible was recon-
structed well. However, we also have to admit that lim-
ited bone height comparing with a dentate mandible is
the main disadvantage of this flap [9,11]. The loss of
vertical bone height results in an unfavorable crown-root
ratio when dental implants are planned for occlusal reha-
bilitation. In this series, we addressed this problem via
vertical DO on the fibula graft.
In the last decade, DO become a popular modality in
correcting craniomaxillofacial bone malformations [12],
especially in managing the mandibular hypoplasia, man-
dibular defect and loss of alveolar ridge height [2,13]. As
far as we know, only a few cases of vertical DO of a fib-
ula flap have been reported in the literature [14-21].
However, the advantages of DO, such as distraction of
soft tissue along with lengthening the bone, the predict-
able outcome, the simplicity of the procedure, the lower
postoperative morbidity without the necessity for bone
grafts or donor sites, determines vertical DO is an excel-
lent and reliable method to increase fibular bone height
according to the individual local needs [13-21]. So we
chose vertical DO on the fibula graft to increase the ridge
volume before placement of dental implants. And the
results showed that the quality of the neogenerated bone
is excellent with adequate characteristics for implant
osseointegration, fully ensuring the following dental im-
plantation and prosthodontic rehabilitation. What is
worth mentioning is the need to wait 6 months after fib-
ula transfer before performing DO. Because during man-
dibular reconstruction, good contour of the fibula usually
required multiple osteotomies, which interrupted the
medullary vessel [14,19]. Long enough interval can en-
sure complete bone regeneration.
5. CONCLUSION
This report presents two patients affected by extensive
mandibular ameloblastoma. Both the patients underwent
radical resection of tumor and simultaneous mandibular
reconstruction by fibula free flap. Vertical DO on the
fibula graft was applied in order to obtain adequate bone
height and to realize placement of dental implants. After
osseointegration, the patient was rehabilitated with ball
attachment denture. The entire course of treatment takes
only about 15 months. After the four-year follow-up,
neither soft tissue related, nor hard tissue, nor implant
related problems were observed. Satisfactory facial
symmetry, chewing and speech functions of the patient
were restored. So this sequential treatment for extensive
mandibular ameloblastoma can obtain an excellent effect
by the shortest time and the lowest economical cost.
Furthermore, the series also can be used to reconstruct
giant mandibular defects caused by different reasons.
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