ORIGINAL CONTRIBUTIONS
Resection of an ameloblastoma in
a pediatric patient and immediate
reconstruction using a combination
of tissue engineering and
costochondral rib graft
A case report
Jeanette Johnson, DDS; Jonathon Jundt, DDS, MD; Issa
Hanna, DDS; Jonathan W. Shum, DDS, MD; Gary
Badger, DDS, MsD; James C. Melville, DDS
A
meloblastoma is an odontogenic tumor
predominantly occurring in patients who
are in their 20s and 30s.1 It is estimated that
approximately 10% to 15% of amelo-
blastomas occur in patients who are younger than age
18 years.1 Resection of larger tumors can leave pa-
tients with continuity defects, poor oral functioning,
and facial deformities. Autogenous cancellous bone
marrow grafts, distraction osteogenesis, or free flaps
are used for immediate reconstruction; however, each
technique has its morbidity and downfalls.2 Surgeons
have reported using a combination of osteo-
conductive (allogeneic bone), osteoinductive (re-
combinant human morphogenetic protein-2
[rhBMP-2]), and osteogenic (bone marrow aspirate)
agents to reconstruct large benign tumor defects in
adults.3,4 In this article, we describe a pediatric patient
who underwent resection of a mandibular unicystic
ameloblastoma with immediate reconstruction using
a costochondral rib graft, allogeneic bone, bone
marrow aspirate concentrate (BMAC), and rhBMP-2.
CASE REPORT
With the approval of the institutional review board
of the University of Texas Health Sciences Center
at Houston, Houston, Texas, we report a case of a
healthy 11-year-old girl who was referred to our
clinic for evaluation of a unilocular radiolucent
lesion of the posterior left mandible.
Copyright ª 2016 American Dental Association. All rights reserved.
ABSTRACT
Background and Overview. Ameloblastoma is an odon-
togenic tumor predominantly occurring in patients who are in
their 20s and 30s. Approximately 10% to 15% of amelo-
blastomas occur in patients younger than 18 years. Although it is
a benign tumor, an ameloblastoma can have a devastating effect
on children both physically and emotionally. The aim of this
case report is to demonstrate how tissue engineering and sur-
gical techniques can minimize morbidity and recovery time
after extirpation and immediate reconstruction of a mandibular
ameloblastoma.
Case Description. An 11-year-old girl was referred for sur-
gical evaluation of a lesion found on a routine dental radiograph.
Resection of a mandibular unicystic ameloblastoma resulted,
including immediate reconstruction using a costochondral rib
graft, allogeneic bone, bone marrow aspirate concentrate, and
recombinant human morphogenetic protein-2. One year post-
operatively, the patient had no evidence of recurrence as well as
excellent mandibular bone height and width with good facial
form. The patient has returned to her daily life without any
disabilities or disfigurement.
Conclusions and Practical Implications. Dentists are
typically the first health care providers to discover oral pathol-
ogy in patients. The coordination of care by the dental care
providers and the oral and maxillofacial specialist was key to the
successful outcome for this patient. With biotechnology and
surgical techniques, the dental surgeon can extirpate an ame-
loblastoma and reconstruct the mandible defect to the ideal
shape and size with minimal morbidity and recovery time.
Key Words. Tissue engineering; oral and maxillofacial sur-
gery; oral and maxillofacial pathology; neoplasms; pediatric
dentistry; oral surgical procedures; bone grafting; bone
marrow transplantation; bone substitutes.
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http://dx.doi.org/10.1016/j.adaj.2016.06.010
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 ORIGINAL CONTRIBUTIONS
Figure 1. Orthopantomograph of a patient with a tooth displaced to-
ward the angle of the mandible, extensive bony resorption, and lingual
displacement of teeth nos. 18 and 19.
Figure 2. Costochondral rib graft secured to custom reconstruction
plate before insertion.
Figure 3. Resected ameloblastoma from the left mandible.
The lesion had been found on a routine dental
radiograph. We obtained a cone-beam computed tomo-
graphic scan, which revealed a 3.4  4.2  3.1-centimeter,
well-defined, osteolytic lesion associated with an
impacted tooth no. 17. The tooth was displaced toward
the angle of the mandible with extensive bony resorption
and lingual displacement of teeth nos. 18 and 19. The
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patient also had marked facial asymmetry and vestibular
expansion of the alveolus (Figure 1); however, neither
erythema, ulceration, nor neurologic deficits were noted.
We performed an endoscopically assisted incisional
biopsy while the patient was under general anesthesia,
which yielded the histologic diagnosis of ameloblastoma.
We created a treatment plan for surgical resection with
immediate reconstruction.
Due to the size of the anticipated defect and desire for
immediate reconstruction, we consulted with the patient
and her family for their consent to the off-label use of
rhBMP-2 and BMAC. We explained all other options in
full, and we made the decision to proceed. We harvested
a costochondral rib graft to reconstruct the mandibular
condyle (Figure 2), and we constructed the 12-cm con-
tinuity defect (Figure 3) with
- a resorbable mesh (poly-[D,L]-lactide co-polymer)
(KLS Martin USA);
- 120 milliliters of allogeneic corticocancellous bone
(Musculoskeletal Transplant Foundation);
- 12 milligrams of bone morphogenic protein (BMP)
(Medtronic);
- 100 mL of BMAC.
The final pathology report revealed unicystic amelo-
blastoma with focal intraluminal plexiform features.
The patient did well postoperatively and remained
in maxillomandibular fixation for 3 weeks. She spent
a total of 2 nights in the pediatrics unit. One year
postoperatively, we provided the patient with an
Essix retainer to act as a removable space maintainer
while she awaits a removable partial denture in the
future. A cone-beam computed tomographic scan at
the 1-year follow-up examination showed adequate bone
with excellent height, width, and arch coordination
for endosseous dental implant placement (Figure 4).
The treatment planned is for the patient to re-
ceive dental implants on reaching skeletal maturity. At
the same time of the implant surgery, the patient also will
have some mandibular recontouring due to excessive
bone formation (Figures 5 and 6).
DISCUSSION
Multiple treatment modalities for ameloblastoma are
discussed in the literature. Common treatment options
include enucleation, curettage, enucleation with chemo-
therapeutics (Carony’s solution), and segmental or
marginal resection.5-7 Though some sporadic reports in
the literature involve marsupialization, this treatment
modality alone is not intended for treatment of locally
aggressive tumors such as ameloblastoma.6 A review of
the literature found a significant recurrence rate in the
ABBREVIATION KEY. BMAC: Bone marrow aspirate
concentrate. BMP: Bone morphogenic protein. rhBMP-2:
Recombinant human morphogenetic protein-2.
 ORIGINAL CONTRIBUTIONS

long term with all treatment modalities except resec-

tion.7,8 This high recurrence rate is attributed to inade-
quate access for instrumentation of larger lesions or
inability to access portions of lesions to apply chemo-
therapeutic agents.8 It is also shown that unicystic ame-
loblastomas are often composed of several histologic
variants and include areas of more aggressive tumor cells
that invade the cystic lining. This finding indicates an
increased proliferation potential and would require a
more aggressive treatment.7,9,10
Recurrence of the lesion in the adolescent patient
in our case would be devastating if it approximated
the skull base or if instrumentation caused a fracture, as
there was insufficient bone for fixation. Due to the size Figure 4. Cone-beam computed tomographic panoramic reconstruction
and location of the lesion in our young patient, the most at 1-year follow-up examination.
predictable treatment was resection and immediate
reconstruction.
Large mandibular defects may be reconstructed with
autogenous bone grafting, allogeneic bone grafting with
BMP, distraction osteogenesis, or a microvascular free
flap; however, each carries with it the morbidity of the
donor site. Autogenous bone grafting remains the stan-
dard for reconstruction of bony defects of the facial
skeleton.11 Such grafts possess osteoinductive, osteogenic,
and osteoconductive properties, yet—for larger defects—
autogenous grafts may lack sufficient volume to
adequately reconstruct a defect.
Microvascular free flaps have been shown to have a
greater than 95% success rate; however, the reported
complication rate is variable and ranges from 9% to
85%.2 For pediatric patients, microvascular free flaps result
in a prolonged hospital stay and introduce the morbidity
of additional surgical sites and the need for rehabil-
itiation.12 Pogrel and colleagues13 study compared vascular
free flaps and autogenous bone grafting for large defects
and found that although the microvascular free flaps were
more successful than autogenous bone grafting (95% and
76%, respectively), these patients stayed an average of 14
additional days in the hospital. Frequently, both micro-
vascular free flaps and autogenous bone grafting recon-
struction require additional augmentation before the sites
can be appropriately used for oral rehabilitation.2 In our
case study, we were able to reconstruct a larger defect—
approximately 12 cm—immediately after resection Figure 5. Photo at 1-year follow-up examination.
without a prolonged hospital stay.
The combination of allogenic bone, BMAC, and BMP
in adults has proved to be a predictable technique for rhBMP-2 is a naturally occurring growth factor shown
immediate reconstruction after resection of benign tu- to induce de novo bone formation. Genetic recombina-
mors.3,4 The combination of a scaffold (allogenic bone), tion in Chinese hamster ovaries allows for the produc-
osteoprogenitor cells (BMAC), and osteoinductive signal tion of large quantities of rhBMP-2 for use in bone
(BMP) fulfill the biological requirement for de novo bone reconstruction.14 There is, however, limited research on
formation. BMAC serves as the richest and most readily use of rhBMP-2 in children.15 In 2008, when the US Food
available source of these bone-forming cells, which and Drug Administration released a black box warning
otherwise would not be present in sufficient quantities, about possible complications of using BMP in close
and is easily harvested and concentrated without signif- proximity to the airway, an additional warning recom-
icant donor site morbidity.14 mended against use in patients with developing

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 ORIGINAL CONTRIBUTIONS
Figure 6. Intraoral photo of the reconstructed mandible.
skeletons.15,16 Such airway complications can be avoided
in the field of maxillofacial surgery with careful use of
BMP in areas in which swelling would not obstruct the
airway. Trials using BMP in children are lacking in both
sample size and longitudinal follow-up. In 2014, Rocque
and colleagues15 performed a literature search that yiel-
ded a greater than expected number of cases in which
BMP was administered in children with similar com-
plications to more traditional techniques when used in
spinal surgery.15 In oral and maxillofacial surgery, the use
of BMP in children has been largely limited to treat-
ment of alveolar cleft grafting, with excellent results.16
Nonvascular allogenic bone grafting using rhBMP-2 and
BMAC may provide alternative reconstructive options in
select patients.
CONCLUSIONS
Although they are benign tumors, ameloblastomas can
have a devastating effect on children, both physically and
emotionally. With modern biotechnology and surgical
techniques, we can extirpate the tumor and reconstruct
the mandible defect to the ideal shape and size with
minimal morbidity and recovery time. Although the
literature demonstrates that recurrence rates for amelo-
blastomas after resection are low, recurrence can occur
up to 33 years after treatment17; long-term follow-up is
necessary to ensure the patient does not require further
treatment. Excellent coordinated care between the Pe-
diatric and Oral and Maxillofacial Surgery departments
at the School of Dentistry, University of Texas at
Houston, was invaluable to the overall successful
outcome of this case. n
Dr. Johnson is a resident, Department of Oral and Maxillofacial Surgery,
School of Dentistry, University of Texas Health Sciences at Houston,
Houston, Texas.
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Dr. Jundt is an assistant professor, Department of Oral and Maxillofacial
Surgery, School of Dentistry, University of Texas Health Sciences at
Houston, Houston, Texas.
Dr. Hanna is an assistant professor, Department of Oral and Maxillofacial
Surgery, School of Dentistry, University of Texas Health Sciences at
Houston, Houston, Texas.
Dr. Shum is an assistant professor, Department of Oral and Maxillofacial
Surgery, School of Dentistry, University of Texas Health Sciences at
Houston, Houston, Texas.
Dr. Badger is an associate professor and the chairman, Department of
Pediatric Dentistry, School of Dentistry, University of Texas Health Sciences
at Houston, Houston, Texas.
Dr. Melville is an assistant professor, Department of Oral and Maxillo-
facial Surgery, School of Dentistry, University of Texas Health Sciences
Center at Houston, 7500 Cambridge St., Houston, Texas 77030, e-mail
James.C.Melville@uth.tmc.edu. Address correspondence to Dr. Melville.
Disclosure. None of the authors reported any disclosures.
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