Radiolucent Lesion of the Posterior Mandible

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CLINICOTHERAPEUTIC CONFERENCE J Oral Maxillofac Surg 67:862-866, 2009 Radiolucent Lesion of the Posterior Mandible Brian Cherry, DMD,* Pushkar Mehra, BDS, DMD,† Vikki Noonan, DMD, DMSc,‡ and Dale Baur, DDS, MD§ Ameloblastic carcinoma is an uncommon, malignant odontogenic tumor with a predilection for occurrence in the posterior mandible. Only a limited number of cases have been reported. We report on a 16-year-old male with ameloblastic carcinoma. This appears to be the first description of ameloblastic carcinoma of the mandible treated with: 1) an initial bony reconstruction using a microvascular free fibular flap and subsequent augmentation with conventional autogenous bone grafts, 2) an initial soft-issue reconstruction using a mi- crovascular free-flap transfer and subsequent replace- ment with keratinized tissue grafts, and 3) rehabilitation using an implant-supported prosthesis. Case Presentation A healthy 16-year-old male was referred to the Department of Oral and Maxillofacial Surgery at Boston University Medical Center (Boston, MA) with a 2-month history of a progressively and rapidly enlarging, painful swelling of his right mandible. Physical examination revealed an expansile mass in the right mandibular angle region (Fig 1A). The mass extended from the right angle region superiorly toward the pinna and temporo- mandibular joint regions, and anteriorly towards the mid-man- dibular body region. There was a deviation of the chin to the contralateral side, and localized areas of hypopigmentation were visible over the right lower face. There was no associated trismus, but there was paresthesia in the right inferior alveolar nerve distribution. An intraoral examination showed a hard, board-like expansion extending toward the midline of the floor of mouth and posteriorly into the lateral pharyngeal area. A plain film radiographic examination showed a large, osteo- lytic, radiolucent lesion of the right mandibular ramus and body, causing significant displacement of an unerupted third molar (Fig 1B). Treatment Recommendations Dale A. Baur, DDS, MD Malignant odontogenic tumors are fortunately a rare occur- rence. In this very interesting case, an incisional biopsy of a rapidly enlarging mandibular lesion in a 16-year-old male dem- onstrated islands of odontogenic epithelium resembling an ameloblastoma. However, there are obvious malignant cyto- logic characteristics in the specimen including sheets of atyp- ical spindled epithelial cells exhibiting nuclear pleomorphism, mitotic figures, multiple nucleoli, and hyperchromaticity. Historically, there are 2 malignant variants of the ameloblas- toma, specifically the malignant ameloblastoma and the amelo- blastic carcinoma. The malignant ameloblastoma is character- ized cytologically by a benign appearance in the primary tumor location, but with documented evidence of distant metastasis. On the other hand, the ameloblastic carcinoma has a histological appearance of an ameloblastoma, but also con- tains cytological malignant features. Based on the photomicro- graphs provided in this case, the diagnosis is most consistent with an ameloblastic carcinoma. In 2005, the World Health Organization provided a classifi- cation of ameloblastic carcinomas. 1 In this scheme, ameloblas- tic carcinomas are subdivided into primary and secondary types. The primary type has the overall histological pattern of ameloblastoma, in addition to malignant cytological patterns. Secondary types, also referred to as dedifferentiated types, arise within a pre-existing ameloblastoma. The secondary type of ameloblastic carcinomas is further subdivided into intraosse- ous and peripheral types. Unlike more common malignancies of the oral cavity and jaws, there is a paucity of literature on the origin, biologic behavior, and treatment of the ameloblastic carcinoma. 2 Avon et al reported in 2003 that the lesion occurs primarily in the mandible, with no gender or race predilection. 3 Clinical pre- sentations range from asymptomatic expansion to those with obvious malignant features, as in this case, with rapid expan- sion, paresthesia, pain, and imaging studies demonstrating extensive bony destruction. The aggressive nature and ten- dency of this tumor to metastasize was noted in this review. Corio et al, in 1987, reported on 8 cases registered at the Armed Forces Institute of Pathology. 4 This case series found a mean age of 30.1 years, with no gender predilection. Seven of these cases involved the mandible. All 8 cases had radio- graphic findings of a poorly defined destructive lesion. One case exhibited lymphatic spread to the neck. Rapid growth, expansion, and trismus were common findings. In this report, Corio et al also noted the tendency of this tumor to have *Former Resident, Department of Oral and Maxillofacial Surgery, Boston Medical Center and Boston University School of Dental Medicine, Boston, MA; Currently, Private Practice, Columbia, SC. †Director and Vice-Chairman, Department of Dentistry and Oral and Maxillofacial Surgery, Boston Medical Center, and Associate Professor and Director of Residency Training, Department of Oral and Maxillofa- cial Surgery, Boston University School of Dental Medicine, Boston, MA. ‡Assistant Professor, Department of Oral and Maxillofacial Pa- thology, Boston University School of Dental Medicine, Boston, MA. §Associate Professor and Chair, Department of Oral and Maxillo- facial Surgery, Case Western Reserve University, Cleveland, OH. Address correspondence and reprint requests to Dr Mehra: De- partment of Oral and Maxillofacial Surgery, Boston University School of Dental Medicine, 100 East Newton Street, Suite G-407, Boston, MA 02118; e-mail: [email protected] © 2009 American Association of Oral and Maxillofacial Surgeons 0278-2391/09/6704-0023$36.00/0 doi:10.1016/j.joms.2008.01.061 862

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CLINICOTHERAPEUTIC CONFERENCE

Oral Maxillofac Surg7:862-866, 2009

Radiolucent Lesion of thePosterior Mandible

Brian Cherry, DMD,* Pushkar Mehra, BDS, DMD,†

Vikki Noonan, DMD, DMSc,‡ and Dale Baur, DDS, MD§

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meloblastic carcinoma is an uncommon, malignantdontogenic tumor with a predilection for occurrence

n the posterior mandible. Only a limited number ofases have been reported. We report on a 16-year-oldale with ameloblastic carcinoma. This appears to be

he first description of ameloblastic carcinoma of theandible treated with: 1) an initial bony reconstruction

sing a microvascular free fibular flap and subsequentugmentation with conventional autogenous bonerafts, 2) an initial soft-issue reconstruction using a mi-rovascular free-flap transfer and subsequent replace-ent with keratinized tissue grafts, and 3) rehabilitation

sing an implant-supported prosthesis.

ase PresentationA healthy 16-year-old male was referred to the Department

f Oral and Maxillofacial Surgery at Boston University Medicalenter (Boston, MA) with a 2-month history of a progressivelynd rapidly enlarging, painful swelling of his right mandible.hysical examination revealed an expansile mass in the rightandibular angle region (Fig 1A). The mass extended from the

ight angle region superiorly toward the pinna and temporo-andibular joint regions, and anteriorly towards the mid-man-

ibular body region. There was a deviation of the chin to theontralateral side, and localized areas of hypopigmentationere visible over the right lower face. There was no associated

rismus, but there was paresthesia in the right inferior alveolarerve distribution. An intraoral examination showed a hard,

*Former Resident, Department of Oral and Maxillofacial Surgery,

oston Medical Center and Boston University School of Dental

edicine, Boston, MA; Currently, Private Practice, Columbia, SC.

†Director and Vice-Chairman, Department of Dentistry and Oral and

axillofacial Surgery, Boston Medical Center, and Associate Professor

nd Director of Residency Training, Department of Oral and Maxillofa-

ial Surgery, Boston University School of Dental Medicine, Boston, MA.

‡Assistant Professor, Department of Oral and Maxillofacial Pa-

hology, Boston University School of Dental Medicine, Boston, MA.

§Associate Professor and Chair, Department of Oral and Maxillo-

acial Surgery, Case Western Reserve University, Cleveland, OH.

Address correspondence and reprint requests to Dr Mehra: De-

artment of Oral and Maxillofacial Surgery, Boston University

chool of Dental Medicine, 100 East Newton Street, Suite G-407,

oston, MA 02118; e-mail: [email protected]

2009 American Association of Oral and Maxillofacial Surgeons

278-2391/09/6704-0023$36.00/0

Coi:10.1016/j.joms.2008.01.061

862

oard-like expansion extending toward the midline of theoor of mouth and posteriorly into the lateral pharyngeal area.plain film radiographic examination showed a large, osteo-

ytic, radiolucent lesion of the right mandibular ramus andody, causing significant displacement of an unerupted thirdolar (Fig 1B).

reatment Recommendations

Dale A. Baur, DDS, MDMalignant odontogenic tumors are fortunately a rare occur-

ence. In this very interesting case, an incisional biopsy of aapidly enlarging mandibular lesion in a 16-year-old male dem-nstrated islands of odontogenic epithelium resembling anmeloblastoma. However, there are obvious malignant cyto-ogic characteristics in the specimen including sheets of atyp-cal spindled epithelial cells exhibiting nuclear pleomorphism,

itotic figures, multiple nucleoli, and hyperchromaticity.Historically, there are 2 malignant variants of the ameloblas-

oma, specifically the malignant ameloblastoma and the amelo-lastic carcinoma. The malignant ameloblastoma is character-

zed cytologically by a benign appearance in the primaryumor location, but with documented evidence of distantetastasis. On the other hand, the ameloblastic carcinoma hashistological appearance of an ameloblastoma, but also con-

ains cytological malignant features. Based on the photomicro-raphs provided in this case, the diagnosis is most consistentith an ameloblastic carcinoma.In 2005, the World Health Organization provided a classifi-

ation of ameloblastic carcinomas.1 In this scheme, ameloblas-ic carcinomas are subdivided into primary and secondaryypes. The primary type has the overall histological pattern ofmeloblastoma, in addition to malignant cytological patterns.econdary types, also referred to as dedifferentiated types,rise within a pre-existing ameloblastoma. The secondary typef ameloblastic carcinomas is further subdivided into intraosse-us and peripheral types.

Unlike more common malignancies of the oral cavity andaws, there is a paucity of literature on the origin, biologicehavior, and treatment of the ameloblastic carcinoma.2 Avont al reported in 2003 that the lesion occurs primarily in theandible, with no gender or race predilection.3 Clinical pre-

entations range from asymptomatic expansion to those withbvious malignant features, as in this case, with rapid expan-ion, paresthesia, pain, and imaging studies demonstratingxtensive bony destruction. The aggressive nature and ten-ency of this tumor to metastasize was noted in this review.orio et al, in 1987, reported on 8 cases registered at thermed Forces Institute of Pathology.4 This case series found aean age of 30.1 years, with no gender predilection. Seven of

hese cases involved the mandible. All 8 cases had radio-raphic findings of a poorly defined destructive lesion. Onease exhibited lymphatic spread to the neck. Rapid growth,xpansion, and trismus were common findings. In this report,

orio et al also noted the tendency of this tumor to have

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requent recurrences. Benlyazid et al reported on a case of aaxillary ameloblastic carcinoma and reviewed the literature

n 2007.5 Of the 65 cases they reviewed in the internationaliterature, they reported a median age of 44 years, with twohirds of the cases occurring in men. The mandible alsohowed a two thirds predominance for location. From theireview, the estimated 5-year survival rate was 68%, with mor-ality generally due to metastatic spread to the lungs, brain, andther bones.

In 2007, Akrish et al presented a comprehensive review ofhe literature and analyzed 37 cases of ameloblastic carcinomaeported from 1984 to 2004.6 In this review, they found a maleo female incidence of almost 2:1. Age at diagnosis rangedrom 15 to 84 years, with a mean age of 52 years. The mandibleas the location of the tumor in 66% of the cases. In this case

eries, common presentations included expansion, pain, toothobility, ulceration, trismus, and paresthesia. Of the 37 cases

eviewed, the survival status was available for 23 cases at theime of the original publication. Fourteen patients were alive,hile 9 had expired. Of the 9 patients that expired, 7 had a

eported recurrent or metastatic tumor. The exact cause ofeath was not determined for all 9 patients, but four patientsere confirmed to have died from metastatic or uncontrolled

ocal/regional recurrence.From the available literature over the last 20 years, one can

onclude that the ameloblastic carcinoma, especially of theandible, is a highly malignant neoplasm with a biological

ehavior necessitating an aggressive surgical approach to pro-

IGURE 1. A, A facial photograph shows significant facial asymme-ry because of an expansile lesion of the right posterior mandible andoor of the mouth. B, A panoramic radiograph shows a large, radiolu-ent lesion of the right mandibular ramus, with a displaced third molar.

herry, Mehra, and Noonan. Radiolucent Lesion of the Posterior Man-ible. J Oral Maxillofac Surg 2009.

ide the best chance of survival.3,7-10 Whether this particularCM

umor arose de novo, or dedifferentiated from an existingmeloblastoma is irrelevant, as my surgical managementould be the same. I feel the work-up already completed in

his case is clinically sufficient to proceed with surgery.As a first step in this case, strong consideration would be

iven to securing the airway with a temporary tracheostomy,hich would be maintained in the early postoperative period.o improve surgical access to the tumor, I would perform a lipplitting incision of the lower lip, extending this incision infe-iorly and posteriorly into the neck as the horizontal compo-ent of a “wine glass” type incision. A vertical componentdded to this incision extending inferiorly to the clavicleould complete surgical access for a neck dissection.The primary tumor should be removed in an en-bloc fash-

on, with 1 to 1.5 cm margins in all dimensions. The adequacyf the soft tissue margins need to be verified by frozen sectionontrol. Oncologically sound bony margins may be verifiedith scrapings of the marrow space in the proximal and distal

egments submitted for frozen section analysis. If the overlyingkin was freely movable and not fixed to the tumor, it woulde maintained, which I would expect to be the case with thisatient. In addition, if not encased in the tumor, I would

IGURE 2. A, Odontogenic epithelial islands show peripheral co-umnar differentiation, with reverse nuclear polarity and central stel-ate-reticulum-like areas (hematoxylin-eosin stain, original magnifica-ion, �100). B, A proliferation of spindle cells shows numeroustypical mitotic figures, nuclear hyperchromaticity, and pleomorphismhematoxylin-eosin stain, original magnification, �200).

herry, Mehra, and Noonan. Radiolucent Lesion of the Posteriorandible. J Oral Maxillofac Surg 2009.

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864 RADIOLUCENT LESION OF THE POSTERIOR MANDIBLE

ttempt to maintain the marginal mandibular branch of theacial nerve. Although the imaging obtained in this case did notemonstrate nodal involvement in the neck, I feel an electiveeck dissection is indicated to address the risk of occult neckisease and improve overall survival. In this case, I would performselective neck dissection, sparing the spinal accessory nerve,

nternal jugular vein, and sternocleidomastoid muscle. It woulde my intention to remove the tumor and neck contents en-bloc.

The condylar neck appears to be free of involvement ac-ording to the imaging report. I would place a reconstructionlate from the condylar stump the symphysis area to restoreontinuity of the remaining mandible. If in order to maintain

IGURE 3. A, An axial computed tomography scan of the mandiblehowing a large, expansile mass in the left mandible region. B, Three-imensional coronal reconstruction image showing extensive boneestruction.

herry, Mehra, and Noonan. Radiolucent Lesion of the Posterior Man-ible. J Oral Maxillofac Surg 2009.

n oncologically sound surgery, the condyle was unable to beCM

reserved, a reconstruction plate with a condylar prosthesis isn option. Consideration could be given for immediate recon-truction of the mandible with a microvascular free fibularaft, although delayed secondary reconstruction with a non-ascularized posterior iliac crest bone graft is a highly accept-ble alternative. If a free flap was not done, and additional softissue was needed to close the oral defect, a pectoralis majorap would be another option.

The role of postoperative radiation treatment is unknownor this malignant tumor, as there are too few cases to make aefinitive recommendation.3 However, in this circumstance,adiation would be a consideration if there were multipleositive nodes in the pathologic examination of the neck. Thisatient would require close postoperative monitoring for lo-al/regional recurrence or metastatic spread. For the first post-perative year, I would see this patient on a monthly basis.eriodic imaging of the head and neck, as well as distant siteseg, chest x-ray) would be necessary to obtain postoperativeaseline studies. A PET scan would also be a useful study tobtain to monitor for recurrence and or metastatic spread.fter the first year, regular periodic assessment would beecessary to monitor for recurrence and/or the developmentf distant disease.

ubsequent Course

An incisional biopsy was performed under local anesthesian the office. A histopathologic examination of the biopsy

IGURE 4. A, An intraoperative view shows augmentation of thetrophic and fractured vascularized free flap, with autogenous,ancellous marrow grafts harvested from the iliac crest. B, A dentalomputed tomography scan shows a successfully augmented freebular graft with radiopaque implant markers.

herry, Mehra, and Noonan. Radiolucent Lesion of the Posteriorandible. J Oral Maxillofac Surg 2009.

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pecimen showed islands of odontogenic epithelium that ex-ibited peripheral columnar epithelial cells, remarkable foralisading and reversed nuclear polarity (Fig 2A). In addition,heets of atypical, spindled epithelial cells exhibited nuclearleomorphism, occasional atypical mitotic figures, multipleucleoli, and hyperchromaticity (Fig 2B). Keratin pearl forma-ion was focally observed, with infiltrative tumor islands ex-ending to the margins of the tissue sections examined. A finaliagnosis of “malignant odontogenic tumor with features ofmeloblastic differentiation” was established. Whether or nothis neoplasm arose in a preexisting ameloblastoma cannot benequivocally determined.Computed tomography (CT) scanning of the maxillofacial

nd neck regions showed a mass measuring 7 � 7 � 6 cmithin the posterior body and angle of the mandible (Fig 3).here was associated cortical disruption, with a displaced

hird molar. The condylar neck showed no evidence of in-olvement. No significant lymphadenopathy was present, andhe largest node was approximately 10 � 5 mm in measure-ent, and was located slightly anterior to the right subman-

ibular gland. Magnetic resonance imaging was obtained forore complete evaluation, and confirmed the absence of

odal metastasis, and a lack of invasion into the soft tissues ofhe floor of the mouth and tongue. A CT examination of thehest and abdominal regions was unremarkable.In view of the rarity and complexity of the tumor, the patientas presented at a combined hospital tumor board for planningf comprehensive treatment. The proposed treatment included aisarticulation segmental resection with 2-cm bony margins, re-ection of the soft tissues to clear margins, a tracheostomy, mod-fied radical neck dissection, and reconstruction of the mandible

ith a fibular, osteomyocutaneous, vascularized free flap, andostoperative radiation therapy. In preparation for surgery, aagnetic resonance angiogram of the lower extremities was

btained, and it showed good 3-vessel runoff.The patient underwent surgery without complications, and

histopathologic examination was consistent with a diagnosisf ameloblastic carcinoma. All bony surgical margins and

ymph nodes were found to be free of tumor. However, theargins were close and less than 2 cm (but greater than 1 cm)

n some areas. The case was discussed at the local hospital’sumor board, and it was decided that additional surgery was notecommended at this stage. The patient had an unremarkableourse in the immediate postoperative period. Radiation therapyas initiated, and the patient received a total dose of 6,000 cGy to

IGURE 5. A panoramic radiograph shows the atrophic (andreviously fractured) free fibular graft after the placement of 5ndosseous dental implants.

herry, Mehra, and Noonan. Radiolucent Lesion of the Posteriorandible. J Oral Maxillofac Surg 2009.

he primary site and the neck in fractionated doses. s

Approximately 1 year after free-flap reconstruction, the patientresented with intermittent swelling of the anterior fibular andandibular area. Clinical and radiographic examinations showedlikely fracture in the anterior part of the microvascular graft.

ome purulent discharge was present around the anterior fibularnd mandibular region. Initially the infection was managed suc-essfully with local debridement in the office, oral antibioticherapy, and 30 dives of hyperbaric oxygen (HBO) therapy. Theatient expressed a desire for prosthetic rehabilitation with en-osseous dental implants. Because previous CT scanning revealedn atrophic fibula and mandible which had already fractured, aecision was made to augment the microvascular fibular graftith iliac crest bone grafts, followed by delayed implant place-ent. The mandibular reconstruction plate was removed, the

rea of the fracture was debrided, and augmentation of the graftas performed as an outpatient surgery procedure, with cortico-

ancellous bone grafts harvested from the anterior iliac crest (Fig). Postoperatively, the patient received 10 additional HBO ses-ions, and healing was uneventful.

Placement of 5 endosseous dental implants was performedpproximately 5 months after grafting (Fig 5). Secondary soft-issue reconstruction was performed 6 months after implantlacement. This included debulking of the excessively thick

ntraoral skin-muscle tissue flap with a modified vestibulo-lasty, using keratinized tissue grafts harvested from the pal-te. The patient was prosthetically rehabilitated with a man-ibular hybrid prosthesis. As of the writing of this report, theatient has recently been diagnosed with metastases to therain and lung regions. He has undergone frontal bone andural resection, reconstruction of the frontal region with aericranial flap, and lower left lobe lung removal. Chemother-py treatment with paclitaxel and carboplatin is continuing.

iscussion

Ameloblastoma is a locally infiltrative, benign, odonto-enic neoplasm typically arising in the posterior regions ofhe jaws. Approximately 80% of ameloblastomas were re-orted to occur in the mandible, and 20% in the maxil-

a.10,11 A rare, malignant variant of ameloblastoma was firstescribed in the medical literature in 1950.12 The Worldealth Organization separated this variant into its current

ubtypes, ie, malignant ameloblastoma and ameloblasticarcinoma, in 1972.13 This classification was further re-ised by Slootweg and Muller in 1984.14 The World Healthrganization described malignant ameloblastoma as a tu-or showing the histopathologic features of classic amelo-lastoma with metastatic deposits, and defined amelo-lastic carcinoma as an epithelial proliferation withistopathologic features of malignancy, either associatedith an ameloblastoma (carcinoma ex-ameloblastoma) or

epresenting a de novo carcinoma that resembles amelo-lastoma histopathologically.15

The histopathologic features of both the primary andetastatic foci of malignant ameloblastoma are indistin-

uishable from conventional ameloblastoma. Thus, metas-ases must be present to establish a diagnosis of malignantmeloblastoma. Metastatic foci were reported in the lung75%), cervical lymph nodes (15%), and spine (15%), andess frequently in the liver, skull, diaphragm, and brain.11

onversely, ameloblastic carcinoma shows the histopatho-ogic features of malignancy, with or without metastatic

pread. This lesion may arise de novo, ex-ameloblastoma,

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866 RADIOLUCENT LESION OF THE POSTERIOR MANDIBLE

r ex-odontogenic cyst,14 and is remarkable for its aggres-ive clinical course. Ameloblastic carcinoma has a predilec-ion for occurrence in the posterior mandible. Only a lim-ted number of cases were reported in the maxilla.10,16

lthough the tumor can present over a wide age range, theean age of patients diagnosed with ameloblastic carci-

oma is 30.1 years. Most patients present with corticalxpansion and pain, and aggressive behavior and recur-ence are frequently cited in the clinical course.4

A diagnosis of ameloblastic carcinoma was established inur patient, based on histopathologic features includingvidence of ameloblastic differentiation together with theresence of nuclear pleomorphism and atypical mitoticgures. The lesion was rapidly growing and locally aggres-ive, with bone destruction, and showed no evidence ofetastases. Wide, local excision with 2-to-3-cm bony mar-

ins is considered the treatment of choice.3,17 Neck dissec-ions are usually recommended. Although postoperativeadiotherapy should be strongly considered, especially forarge lesions, there is a paucity of well-documented cases toatify this protocol.16,18,19

Our patient had a composite resection of the hemiman-ible via combined extraoral and intraoral approaches.lthough the condylar neck was spared from the tumor, a

emporomandibular joint disarticulation resection wasompleted, sparing the meniscus, because the requiredurgical margins necessitated condylar sacrifice. Meniscalreservation was previously reported during disarticulationesections in the treatment of malignancies that spared theondylar neck region.20 A microvascular, osteocutaneous,bular free flap was initially used to reconstruct the result-nt hard-tissue and soft-tissue defects after tumor extirpa-ion, because the patient preferred one-stage treatmentbone/soft-tissue resection, and concomitant reconstruc-ion), compared with the conventional staged treatmentith pedicled myocutaneous flaps and delayed bone graft-

ng. In addition, reconstruction of the mandible with bonen the form of a free flap allowed for immediate postoper-tive radiation treatment, which was strongly recom-ended by the hospital’s tumor board, given the “close”

oft-tissue margins. The development of a delayed fracturet the collapsed osteotomy site of the fibular graft wasuccessfully treated with local debridement, antibiotics,BO therapy, and simultaneous grafting with iliac-crestone grafts in preparation for dental implants. All implantssseointegrated, and were restored without complications.econdary soft-tissue reconstruction with keratinized graftsas unremarkable.Ameloblastic carcinoma has reported recurrence rates

f between 15%19 and 60%,17 even when treated withggressive surgical excision. Five-year survival rates wereeported to be less than 40%.4,17 Because of recurrencend reported metastases to the lungs and lymphodes,3,4,14,18,21 close, periodic clinical and radiographice-evaluation of the patient is mandatory. Our patient haseen recently diagnosed with metastatic lesions in the

rain and lung.

This appears to be the first report of ameloblastic carci-oma of the mandible treated with: 1) an initial bonyeconstruction using a microvascular free fibular flap andubsequent augmentation with conventional autogenousone grafts, 2) an initial soft-issue reconstruction usingicrovascular free-flap transfer and subsequent replace-ent with keratinized tissue grafts, and 3) complete, suc-

essful, dental prosthetic rehabilitation, using an implant-upported prosthesis in a radiated and reconstructedandible after HBO therapy. However, despite these ef-

orts and “presumably” adequate treatment, long-termrognosis for such patients remains poor.

eferences1. Barnes L, Eveson JW, Reichart P, et al (eds): Pathology and

Genetics: Head and Neck Tumors. Lyon, World Health Organi-zation, 2005, pp 287-289.

2. Ward BB, Edlund S, Sciubba J, et al: Ameloblastic carcinoma(primary type) isolated in the anterior maxilla: Case report withreview of the literature. J Oral Maxillofac Surg 65:1800, 2007

3. Avon SL, McComb J, Clokie C: Ameloblastic carcinoma:Case report and literature review. J Can Dent Assoc 69:573, 2003

4. Corio RL, Goldblatt LI, Edwards PA, et al: Ameloblastic carci-noma: A clinicopathologic study and assessment of eight cases.Oral Surg Oral Med Oral Pathol 64:570, 1987

5. Benlyazid A, Lacroix-Triki M, Aziza R, et al: Ameloblastic carci-noma of the maxilla: case report and review of the literature. OralSurg Oral Med Oral Pathol Oral Radiol Endod 104:e17, 2007

6. Akrish S, Buchner A, Shoshani Y, et al: Ameloblastic carcinoma:Report of a new case, literature review, and comparison toameloblastoma. J Oral Maxillofac Surg 65:777, 2007

7. Hall JM, Weathers DR, Unni KK: Ameloblastic carcinoma: Ananalysis of 14 cases. Oral Surg Oral Med Oral Pathol Oral RadiolEndod 103:799, 2007

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9. Goldenberg D, Sciubba J, Koch W, et al: Malignant odontogenictumors: A 22-year experience. Laryngoscope 114:1770, 2004

0. Sastre J, Munoz M, Naval L, et al: Ameloblastic carcinoma of themaxilla: Report of a case. J Oral Maxillofac Surg 60:102, 2002

1. Verneuil A, Sapp P, Huang C, et al: Malignant ameloblastoma:Classification, diagnostic, and therapeutic challenges. Am JOtolaryngol 23:44, 2002

2. Thoma K: Oral Pathology. St Louis, MO, CV Mosby, 1950,pp 1270-1333

3. Pindborg J, Kramer I, Torloni H: Historical Typing of Odonto-genic Tumors, Jaw Cysts, and Allied Lesions. Geneva, Switzer-land, World Health Organization, 1972, pp 35-36

4. Slootweg PJ, Muller H: Malignant ameloblastoma or ameloblasticcarcinoma. Oral Surg Oral Med Oral Pathol 57:168, 1984

5. World Health Organization: World Health Organization Clas-sification of Tumours: Pathology and Genetics of Head andNeck Tumours. Lyon, France, International Agency for Re-search on Cancer Press, 2005

6. Dhir K, Sciubba J, Tufano RP: Ameloblastic carcinoma of themaxilla. Oral Oncol 39:736, 2003

7. Marx RE, Stern D: Oral and Maxillofacial Pathlogy: A Rationalefor Diagnosis and Treatment. Chicago, IL, Quintessence Pub-lishing Co, Inc, 2003, p 657

8. Bruce RA, Jackson IT: Ameloblastic carcinoma. Report of anaggressive case and review of the literature. J CraniomaxillofacSurg 19:267, 1991

9. Datta R, Winston JS, Diaz-Reyes G, et al: Ameloblastic carci-noma: Report of an aggressive case with multiple bony metas-tases. Am J Otolaryngol 24:64, 2003

0. Carlson ER: Disarticulation resections of the mandible: A prospec-tive review of 16 cases. J Oral Maxillofac Surg 60:176, 2002

1. Cizmecy O, Aslan A, Onel D, et al: Ameloblastic carcinoma ex

ameloblastoma of the mandible: Case report. Otolaryngol HeadNeck Surg 130:633, 2004