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Original Investigation |

Tumors of the Pediatric Maxillofacial Skeleton A 20-Year Clinical Study FREE

Katherine S. Perry, MD1; Andrew T. Tkaczuk, MD1; John F. Caccamese Jr, DMD, MD2; Robert A. Ord, DDS, MD2; Kevin D. Pereira, MD, MS1
[+] Author Affiliations
1University of Maryland School of Medicine, Baltimore
2University of Maryland School of Dentistry, Baltimore
JAMA Otolaryngol Head Neck Surg. 2015;141(1):40-44. doi:10.1001/jamaoto.2014.2895.
Text Size: A A A
Published online

Importance  Pediatric jaw tumors are a rare clinical entity and are not well addressed in the otolaryngology literature. It is important that otolaryngologists be familiar with the clinical features, management, and outcomes associated with these lesions.

Objective  To review the clinical presentation, management, and outcomes of jaw tumors in children treated at a tertiary care academic center.

Design, Setting, and Participants  Retrospective medical record review of children 16 years or younger who presented to the departments of Oral-Maxillofacial Surgery and Otorhinolaryngology at the University of Maryland Medical Center between 1992 and 2012 and were diagnosed as having a jaw tumor. A PubMed review of literature from 1992 to 2013 on jaw tumors in children was also conducted.

Main Outcomes and Measures  Medical records were reviewed for data on symptoms, physical findings, pathologic diagnosis, intervention, and outcomes.

Results  The medical records of 76 patients evaluated for a jaw mass were reviewed, and 20 were found to have a diagnosis of a jaw tumor. The 2 most common pathologic diagnoses were ameloblastoma (n = 5) and juvenile ossifying fibroma (n = 4). Two tumors were malignant, a rhabdomyosarcoma and a teratoma. Thirteen patients presented with evidence of a mass or swelling, 5 patients were asymptomatic with a lesion found on surveillance panoramic radiography, and 1 patient presented with epistaxis and 1 with facial weakness and pain. All tumors excluding a lymphangioma and a rhabdomyosarcoma were managed surgically. Eight patients underwent more than 1 procedure including secondary reconstruction prior to a satisfactory outcome.

Conclusions and Relevance  Pediatric jaw tumors are rare lesions most commonly presenting with a swelling or mass. Patients can be asymptomatic with the lesion identified on routine imaging. Certain clinical features such as age, location of tumor, and presence or absence of bone and soft tissue can narrow the differential diagnosis and identify tumors that may be malignant. Incisional biopsy is an important first step. A majority of jaw tumors are benign but require surgical intervention for eradication of disease. Multiple procedures, including reconstruction, may be required for certain lesions prior to cure.

Tumors of the pediatric maxillofacial skeleton are a rare clinical entity with a broad differential diagnosis.1,2 Tumors may be classified by origin as being either odontogenic or nonodontogenic or malignant or benign.3 Odontogenic tumors arise from quiescent tooth-forming tissues of the jaws and are most commonly located within the bones of the jaw (central odontogenic tumor), though occasionally they may arise in surrounding soft tissue (peripheral odontogenic tumor).3 The etiology of these tumors remains unknown, and they vary widely in their level of aggression.3 Nonodontogenic tumors encompass a wide range of pathologic conditions and may arise from mesenchymal tissue within the jaw or from the osseous tissue of the jaw.4 No matter the origin, prompt identification and treatment of jaw tumors is critical because some tumors may be either locally destructive or malignant.3,5 While many jaw masses are discovered by dentists and further managed by oral-maxillofacial surgeons, a patient with a jaw mass may first present to an otolaryngologist for management. Hence, it is important that the clinician understands the clinical features, workup, management, and outcomes associated with these lesions.

A PubMed review of literature from 1992 to 2013 on jaw tumors in children revealed many case reports and series describing children with specific pathologic diagnosis. However, there were very few case series or reviews discussing children with jaw tumors as a whole, and no large series were found within the otolaryngology literature. Many of the larger case series identified in our literature search included oral tumors in addition to those of the jaw, and all differed in their definition of the pediatric population. None of these studies address the clinical presentation of jaw tumors.

Our 20-year experience with these pediatric jaw tumors along with a pertinent review of literature may help the otolaryngologist–head and neck surgeon with early identification and management of these uncommon lesions.

The institutional review board of the University of Maryland, Baltimore, approved this study. The medical records of patients who presented to the departments of Oral-Maxillofacial Surgery and Otorhinolaryngology at the University of Maryland Medical Center from 1992 to 2012 with chief complaints of a jaw mass, swelling, lesion found on panoramic radiography, or jaw pain were reviewed. Of these, the records of 76 patients 16 years and younger at initial presentation were selected for detailed clinical data analysis. Only patients who were diagnosed as having a tumor of the maxilla or mandible were included in this study. Medical records were reviewed for symptoms, physical examination findings, radiological findings, pathologic diagnosis, intervention, and outcomes.

Twenty children had a diagnosis consistent with a jaw tumor. A total of 12 different pathologic diagnoses were identified (Table 1). Thirteen patients presented with evidence of a mass or swelling, 5 patients were asymptomatic with a lesion found on surveillance panoramic radiography, and 1 patient presented with epistaxis and 1 with facial weakness and pain. There were 2 malignant tumors, a rhabdomyosarcoma and a teratoma. The 2 most common diagnoses were ameloblastoma (n = 5) and juvenile ossifying fibroma (JOF) (n = 4). Eleven tumors were in the mandible and 9 in the maxilla. The ages at presentation ranged from 8 months to 16 years. Four patients were in the 0- to 5-year age group, 6 were in the 6- to 10-year age group, and 10 were in the 11- to 16-year age group. Of the children with jaw tumors, 11 were female and 9 were male. Ten tumors were odontogenic and 10 were nonodontogenic in origin. The presenting symptoms, ages at presentation, location of the lesion, and number of surgical procedures required for successful excision of the tumors are given in Table 2.

Table Graphic Jump LocationTable 1.  Type and Site of Jaw Tumors in the Study
Table Graphic Jump LocationTable 2.  Data Summary for 20 Patients With Jaw Tumors

All patients, excluding 1 infant with suspected lymphangioma and 1 with rhabdomyosarcoma, were treated surgically for their disease. Incisional biopsy was performed for diagnosis prior to definitive treatment in the majority of cases. Four patients were initially treated with enucleation as opposed to incisional biopsy: 3 were suspected of having an odontoma, and 1 exhibited a focus of ameloblastoma within a dentigerous cyst. For all 4 patients, this was the sole procedure. Eight patients underwent more than 1 procedure after initial biopsy, including secondary reconstruction, prior to a satisfactory outcome. Ameloblastomas located in the mandible were treated with segmental resection and reconstruction using a fibula free flap in 1 case and reconstruction bar placement in the rest. Bone grafting was subsequently performed for all patients with a reconstruction bar. The maxillary ameloblastoma was managed by enucleation and did not require reconstruction. The ossifying fibromas (OFs), both juvenile and classic, were treated with enucleation. Two JOFs recurred, one requiring repeated enucleation and another requiring hemimaxillectomy. Fibromas and odontomas were all definitively treated with enucleation. The osteoblastoma was treated with segmental resection leading to resolution. The patient with rhabdomyosarcoma required additional trips to the operating room for postbiopsy hemostasis and was definitively treated with chemotherapy and radiation. The teratoma was treated with wide local excision with calvarial bone graft and a titanium mesh for reconstruction of the right orbital floor. This recurred twice, first requiring endoscopic excision and chemotherapy and next, revision maxillectomy and proton beam radiation therapy.

Jaw tumors in children are relatively uncommon. The exact incidence is hard to estimate owing to the different tumor types and sites included in studies and the differing definitions used by authors to define the pediatric age group. Of the large studies reviewing jaw tumors that we identified in our literature search, 4 include oral cavity tumors and 1 includes only odontogenic tumors, both of which differ from our study.15 Tumors of the jaw have a broad differential diagnosis including both benign and malignant lesions, which may originate from bone or soft tissue and may be odontogenic or nonodontogenic. The incidence of nonodontogenic tumors is twice that of odontogenic ones.

The most common diagnoses in our series, ameloblastoma and JOF, were also the most common tumors in similar studies.1,2 Similarly, odontoma has also been found to be a common diagnosis, though our series had only 2 cases.2,6 The lower incidence in our study may be due to our small sample size or that odontomas are easily identified and commonly managed outside of the tertiary centers. Of our 20 patients with jaw tumors, 90% were benign and 10% were malignant. Reported rates of malignancy in similar series were 2.5%, 2.9%, and 7%.1,2,6 There were no neonatal cases in this series because of the referral pattern for our institution as well as the extremely low incidence of these tumors in that age group.

The majority of patients presented with an asymptomatic lesion found on panoramic radiography or with a jaw mass. According to our data, the chief complaint of a jaw mass was more often associated with an ameloblastoma or JOF, making odontoma less likely. Presentation with an asymptomatic lesion found on panoramic radiography without a palpable mass made the first 2 diagnoses less likely, although on occasion an ameloblastoma can present this way. Because ameloblastoma and JOF are relatively fast-growing tumors compared with their other benign counterparts, they are likely to present with a mass shortly after development. Odontomas are very slow growing and therefore not clinically apparent in their early stages but are picked up when the patient undergoes a screening panoramic radiography. The number of tumors identified incidentally on panoramic radiography highlights the importance of routine dental surveillance in children. Pain was present in 8 cases but did not correlate with pathologic diagnosis in our study. Nonetheless, both malignant tumors in our series did present with pain. Additional symptoms were epistaxis associated with the rhabdomyosarcoma and facial weakness associated with the teratoma. Bleeding from a tumor and facial weakness should be considered ominous signs. When our data were broken down by age, we found a difference in the distribution of diagnoses. In children younger than 11 years, JOF was the most common diagnosis and only 1 ameloblastoma was seen. Both malignant lesions were seen in this age group. In children 11 years and older, ameloblastoma was the most common diagnosis, with odontoma and JOF being common as well. These numbers are supported by the fact that ameloblastomas are known to be rare in children younger than 11 years. Two studies found only 10.8% of ameloblastomas in children younger than 11 years.5,7 Our patients with ameloblastoma were 8, 11, 13, 14, and 15 years old, which is consistent with the age range sited in the literature. When evaluating a child with a new jaw mass, keeping this age distribution in mind will be helpful in narrowing the differential diagnoses. When the data were analyzed by location, we found ameloblastomas to be predominantly localized to the mandible and JOF to the maxilla. This is consistent with the reported site predilection of these lesions.

After identification of a jaw mass, workup in most cases began with an incisional biopsy prior to attempting a definitive procedure. Because management varies for different pathologic diagnoses, performing a biopsy prior to a definitive procedure is useful. The exceptions in our study were 3 patients suspected of having odontomas that were managed with enucleation without preceding incisional biopsy. While frozen section can be used to diagnose tumors of the jaws with comparable accuracy to incisional biopsy, the diagnostic accuracy of frozen section for jaw tumors differed by 10%.8 This could result in the overtreatment of some tumors in this population in which the aesthetic and growth implications of treatment can be profound. In our experience, most jaw lesions can be readily biopsied in the office with local anesthesia or moderate sedation with little additional inconvenience to the family. In addition, should the tumor require continuity resection of the mandible or maxillectomy, an appropriate reconstructive strategy can be planned for while undertaking definitive treatment, whether that involves fabrication of an obturator, precontouring a reconstruction plate, or planning a free tissue transfer.

Reconstruction was required for all mandibular lesions requiring wide excision and for 1 maxillary lesion. Reconstruction options include reconstruction bar with iliac crest bone graft or free flap reconstruction, both of which were used in our population. The decision to use nonvascularized graft vs free tissue is made based on the size of the defect and the location of the defect, as well as the extent of the resection or the need for adjuvant therapy. When the periosteum is resected as a margin, the wound bed becomes more hostile for bone regeneration as well as for nonvascularized graft healing. For larger defects of this nature, composite free tissue transfer is a good option. Additional procedures were performed for recurrence, dental implants, cosmetic revision, second look, and repeated biopsy.

Ameloblastoma is a benign odontogenic tumor most frequently found in adolescents when of the unicystic variety. When multicystic, they are seen most frequently in young adults in the third and fourth decade if life.5 While some sources cite ameloblastoma as the most common benign odontogenic tumor,5 others believe that odontoma merits that distinction.9 Despite being benign, ameloblastomas are locally aggressive tumors with a recurrence rate of as high as 75% to 90% for solid tumors treated with enucleation.5 Therefore, wide resection with partial mandibulectomy or maxillectomy is often required to decrease the likelihood of recurrence. Our study identified 5 patients with ameloblastomas: 4 in the mandible and 1 in the maxilla. Ameloblastomas located in the mandible were all treated with segmental resection with reconstruction using fibula free flap in 1 case and reconstruction bar placement in 3 cases. Bone grafting was subsequently performed for all 3 patients with a reconstruction bar. Scar revision was required in 1 case, in which the maxillary ameloblastoma was identified as a focus of disease within a dentigerous cyst. Because the initial diagnosis was a dentigerous cyst, this patient was treated with enucleation and did not require reconstruction. Despite high reported recurrence rates, no patients in our study had recurrence after resection of ameloblastoma. This is likely a reflection of the fact that our patients with ameloblastoma were managed with incisional biopsy for diagnosis followed by wide excision to eradicate disease.

Juvenile OF is a type of benign fibro-osseous tumor consisting of bands of osseous material within a fibrous stroma.10 It is a distinct subtype of OF that lacks the osteoblastic lining commonly seen in OF10 and is usually only seen up to age 15 years.11 Compared with classic OF, JOF tends to have infiltrative margins leading to a higher risk of recurrence. Juvenile OF is typically found in the paranasal sinuses,10,11 making the mandible a rare location. In keeping with previous observations, all 4 of our cases of JOF were located in the maxilla. Juvenile OF may be treated with enucleation and curettage or wide local excision but may recur with enucleation. In our population, 1 patient had recurrence after enucleation requiring repeated enucleation. Another patient had recurrence after initial wide local excision requiring a hemimaxillectomy.

Odontomas are benign odontogenic tumors composed of enamel and dentin12 and are most commonly seen at age 10 to 19 years.9 Odontomas are characteristically slow growing and are therefore often diagnosed by panoramic radiographic findings of a radiopaque mass near a tooth. In our series, both odontomas were identified on routine panoramic radiography. Based on the characteristic appearance on the panoramic radiograph, a clinical diagnosis of odontoma may be made, and as done in our series, one may proceed to enucleation as the initial procedure.10,12

The purpose of this study was to alert the practitioner unfamiliar with pediatric jaw pathology to the possible diagnoses encountered when treating childhood jaw tumors. Because of the variety of lesions that one can expect to be encountered, their vastly different behavior, and propensity for recurrence, it is difficult to construct an algorithm or treatment pathway. The clinical signs described in this article are pointed out to alert the practitioner to develop a level of concern when encountering them. The recommendations are based on our experience, and we believe that each tumor or lesion should be approached uniquely and treatment should be based on findings from an appropriate preceding tissue biopsy.

Pediatric jaw tumors are rare lesions whose clinical presentation and incidence have not been well described in the otolaryngology literature. These lesions most frequently present either with a palpable jaw mass or with identification of a mass on routine imaging. Certain clinical features such as patient age, location of tumor, and presence or absence of bone and soft tissue can narrow the differential diagnosis. After obtaining a history and results from a physical examination, performing an incisional biopsy should be the next step and will guide further treatment. A majority of jaw tumors are benign but nevertheless require surgical intervention for eradication of disease. Multiple procedures, including advanced reconstructive procedures, may be required for certain lesions prior to cure.

Submitted for Publication: June 4, 2014; final revision received September 22, 2014; accepted October 1, 2014.

Corresponding Author: Kevin D. Pereira, MD, MS, University of Maryland School of Medicine, Department of Otorhinolaryngology, 16 S Eutaw St, Ste 500, Baltimore, MD 21037 (KPereira@smail.umaryland.edu).

Published Online: November 13, 2014. doi:10.1001/jamaoto.2014.2895.

Author Contributions: Drs Perry and Tkaczuk had full access to all of the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis.

Study concept and design: Perry, Pereira.

Acquisition, analysis, or interpretation of data: All authors.

Drafting of the manuscript: Perry, Tkaczuk, Pereira.

Critical revision of the manuscript for important intellectual content: Perry, Caccamese, Ord, Pereira.

Administrative, technical, or material support: Perry, Ord, Pereira.

Study supervision: Perry, Caccamese, Pereira.

Conflict of Interest Disclosures: None reported.

Previous Presentation: These data were presented as a poster at the 2013 American Academy of Otolaryngology–Head and Neck Surgery Annual Meeting; September 29–October 2, 2013; Vancouver, British Columbia, Canada.

Aregbesola  SB, Ugboko  VI, Akinwande  JA, Arole  GF, Fagade  OO.  Orofacial tumours in suburban Nigerian children and adolescents. Br J Oral Maxillofac Surg. 2005;43(3):226-231.
PubMed   |  Link to Article
Sato  M, Tanaka  N, Sato  T, Amagasa  T.  Oral and maxillofacial tumours in children: a review. Br J Oral Maxillofac Surg. 1997;35(2):92-95.
PubMed   |  Link to Article
Mamabolo  M, Noffke  C, Raubenheimer  E.  Odontogenic tumours manifesting in the first two decades of life in a rural African population sample: a 26 year retrospective analysis. Dentomaxillofac Radiol. 2011;40(6):331-337.
PubMed   |  Link to Article
McCarthy  EF.  Fibro-osseous lesions of the maxillofacial bones. Head Neck Pathol. 2013;7(1):5-10.
PubMed   |  Link to Article
Zhang  J, Gu  Z, Jiang  L,  et al.  Ameloblastoma in children and adolescents. Br J Oral Maxillofac Surg. 2010;48(7):549-554.
PubMed   |  Link to Article
Tanaka  N, Murata  A, Yamaguchi  A, Kohama  G.  Clinical features and management of oral and maxillofacial tumors in children. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999;88(1):11-15.
PubMed   |  Link to Article
Chawla  R, Ramalingam  K, Sarkar  A, Muddiah  S.  Ninety-one cases of ameloblastoma in an Indian population: a comprehensive review. J Nat Sci Biol Med. 2013;4(2):310-315.
PubMed   |  Link to Article
Guthrie  D, Peacock  ZS, Sadow  P, Dodson  TB, August  M.  Preoperative incisional and intraoperative frozen section biopsy techniques have comparable accuracy in the diagnosis of benign intraosseous jaw pathology. J Oral Maxillofac Surg. 2012;70(11):2566-2572.
PubMed   |  Link to Article
Soluk Tekkesin  M, Pehlivan  S, Olgac  V, Aksakallı  N, Alatlı  C.  Clinical and histopathological investigation of odontomas: review of the literature and presentation of 160 cases. J Oral Maxillofac Surg. 2012;70(6):1358-1361.
PubMed   |  Link to Article
Triantafillidou  K, Venetis  G, Karakinaris  G, Iordanidis  F.  Ossifying fibroma of the jaws: a clinical study of 14 cases and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;114(2):193-199.
PubMed   |  Link to Article
Kramer  IR, Pindborg  JJ, Shear  M.  The World Health Organization histological typing of odontogenic tumours: introducing the second edition. Eur J Cancer B Oral Oncol. 1993;29B(3):169-171.
PubMed   |  Link to Article
Bordini  J  Jr, Contar  CM, Sarot  JR, Fernandes  A, Machado  MÂN.  Multiple compound odontomas in the jaw: case report and analysis of the literature. J Oral Maxillofac Surg. 2008;66(12):2617-2620.
PubMed   |  Link to Article

Figures

Tables

Table Graphic Jump LocationTable 2.  Data Summary for 20 Patients With Jaw Tumors
Table Graphic Jump LocationTable 1.  Type and Site of Jaw Tumors in the Study

References

Aregbesola  SB, Ugboko  VI, Akinwande  JA, Arole  GF, Fagade  OO.  Orofacial tumours in suburban Nigerian children and adolescents. Br J Oral Maxillofac Surg. 2005;43(3):226-231.
PubMed   |  Link to Article
Sato  M, Tanaka  N, Sato  T, Amagasa  T.  Oral and maxillofacial tumours in children: a review. Br J Oral Maxillofac Surg. 1997;35(2):92-95.
PubMed   |  Link to Article
Mamabolo  M, Noffke  C, Raubenheimer  E.  Odontogenic tumours manifesting in the first two decades of life in a rural African population sample: a 26 year retrospective analysis. Dentomaxillofac Radiol. 2011;40(6):331-337.
PubMed   |  Link to Article
McCarthy  EF.  Fibro-osseous lesions of the maxillofacial bones. Head Neck Pathol. 2013;7(1):5-10.
PubMed   |  Link to Article
Zhang  J, Gu  Z, Jiang  L,  et al.  Ameloblastoma in children and adolescents. Br J Oral Maxillofac Surg. 2010;48(7):549-554.
PubMed   |  Link to Article
Tanaka  N, Murata  A, Yamaguchi  A, Kohama  G.  Clinical features and management of oral and maxillofacial tumors in children. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 1999;88(1):11-15.
PubMed   |  Link to Article
Chawla  R, Ramalingam  K, Sarkar  A, Muddiah  S.  Ninety-one cases of ameloblastoma in an Indian population: a comprehensive review. J Nat Sci Biol Med. 2013;4(2):310-315.
PubMed   |  Link to Article
Guthrie  D, Peacock  ZS, Sadow  P, Dodson  TB, August  M.  Preoperative incisional and intraoperative frozen section biopsy techniques have comparable accuracy in the diagnosis of benign intraosseous jaw pathology. J Oral Maxillofac Surg. 2012;70(11):2566-2572.
PubMed   |  Link to Article
Soluk Tekkesin  M, Pehlivan  S, Olgac  V, Aksakallı  N, Alatlı  C.  Clinical and histopathological investigation of odontomas: review of the literature and presentation of 160 cases. J Oral Maxillofac Surg. 2012;70(6):1358-1361.
PubMed   |  Link to Article
Triantafillidou  K, Venetis  G, Karakinaris  G, Iordanidis  F.  Ossifying fibroma of the jaws: a clinical study of 14 cases and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;114(2):193-199.
PubMed   |  Link to Article
Kramer  IR, Pindborg  JJ, Shear  M.  The World Health Organization histological typing of odontogenic tumours: introducing the second edition. Eur J Cancer B Oral Oncol. 1993;29B(3):169-171.
PubMed   |  Link to Article
Bordini  J  Jr, Contar  CM, Sarot  JR, Fernandes  A, Machado  MÂN.  Multiple compound odontomas in the jaw: case report and analysis of the literature. J Oral Maxillofac Surg. 2008;66(12):2617-2620.
PubMed   |  Link to Article

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