Ameloblastoma maxilar superior: presentación de caso clínico / Upper maxillary ameloblastoma: report of a clinical case

El ameloblastoma es una entidad patológica benigna de origen odontogénico, de alta agresividad local, lo que ha sido un punto de controversia en la literatura científica. Se presenta un caso clínico, con ubicación en maxilar superior con diagnóstico de ameloblastoma sólido multiquístico. Se evalúa el caso, su posibilidad de tratamiento, revisando las características clínicas y anatomopatológicas de laentidad, basándose en una búsqueda bibliográfica.

Ameloblastoma is a benign pathological entity of odontogenic origin, very aggressive at a local level, which has become a controversial issuein cientific literature. A clinic case is presented, located in the superior maxilla diagnosed as a solid multicystic ameloblastoma. The case isevaluated, its ways of treatment, the clinical and anatomopathological characteristics of the entity with an intensive bibliographical researc.

Epidemiologia das lesões fibro-ósseas benignas dos maxilares / Epidemiological study of the benign fibro-osseous lesions (BFOLs) of the jaws

As lesões fibro-ósseas benignas (LFOBs) são condições raras caracterizadas pelo desenvolvimento de doenças neoplásicas, reativas ou displásicas, nas quais o osso normal é substituído por tecido conjuntivo composto por colágeno, fibroblastos e diferentes materiais mineralizados semelhantes ao osso ou cemento. Os principais tipos são: displasia fibrosa (DF): monostótica (DFM) ou poliostótica (DFP); displasia óssea (DO): periapical (DOP), focal (DOFoc) ou florida (DOFlor); e o fibroma ossificante (FO): convencional (FOC) ou juvenil (FOJ). O objetivo deste estudo foi descrever o perfil epidemiológico das LFOBs dos maxilares diagnosticados no serviço de referência em Patologia, Estomatologia e Radiologia da Faculdade de Odontologia da Universidade Federal de Minas Gerais e comparar com os resultados encontrados em outros estudos. Os dados foram obtidos dos arquivos dos serviços clínico e laboratorial, no período de 26 anos (1990-2015). Informações de gênero, idade, raça dos pacientes e principais características clínicas e radiográficas da lesão foram coletadas. Foi realizada a análise estatística descritiva das variáveis e teste de associação entre os grupos de lesões e o gênero, faixa etária, aumento de volume e osteomielite. Entre todos os diagnósticos do período avaliado (27998 prontuários), as LFOBs representaram 1,36% (n=383). As DOs (n=187, 48,8%) foram as mais frequentes, seguidas pela DF (n=103, 26,9%) e FO (n=93, 24,3%). Quanto aos subtipos de LFOBs, as mais frequentes foram a DOFlor e a DFM, ambas com 101 casos (26,4%). As mulheres foram a maioria no estudo, com frequência de 82,0% (n=314), assim como os pacientes não negros (59,0%, n=226). Entre todas as LFOBs a média de idade foi de 38,52 ± 17,54, acometendo mais a 4ª e 5ª décadas. As características radiográficas mais comuns foram imagens mistas: radiopaca e radiolúcida (51,7%). A mandíbula foi a região mais acometida (n=247, 64,5%), exceto para DF que envolveu mais a maxila (n=68, 66,0%). A DF foi observada mais entre os homens que em mulheres, assim como entre os pacientes mais jovens. DOs foram mais comuns em mulheres e em pacientes mais velhos. Enquanto que no FO não foi observada diferença significativa entre o gênero. A associação entre a presença de osteomielite e DO foi estatisticamente significante (p=0,0001). O aumento de volume foi significante na DF e FO (p=0,0001). A frequência de LFOB é semelhante à previamente reportada na literatura no mesmo país, mas difere dos dados observados em outras populações. É importante considerar a correlação de dados clínicos, radiográficos e histopatológico para o diagnóstico definitivo de LFOBs. Estudos epidemiólogicos podem orientar o clínico sobre a perfil do paciente acometido por uma doença. Nossos resultados são importantes para auxiliar no diagnóstico e manejo clínico das lesões fibro-ósseas benignas

Benign fibro-osseous lesions (BFOLs) are rare conditions characterized by the development of neoplastic, reactive or dysplastic disease, in which is observed replacement of the normal bone by collagen, fibroblasts and different mineralized materials like bone and cementum. The main types are: fibrous dysplasia (FD): monostotic (MFD) or polyostotic (PFD); osseous dysplasia (OD): periapical (POD), focal (FocOD) or florid (FlorOD); and ossifying fibroma (OF): conventional (COF) or juvenile (JOF). The aim of this study was to describe the epidemiological profile of BFOLs of the jaws diagnosed in the reference service in oral pathology at the Universidade Federal of Minas Gerais and compare it with the results found in other studies. Data were obtained from the archives of clinical and laboratory services in the period of 26 years (1990-2015). Gender, age, race and main clinical and radiographic characteristics of the lesion were collected. Descriptive statistical analysis of each variable and associations test between groups of lesions and gender, age, swelling and osteomyelitis were made. Among all diagnoses in the period (n=27998 records), the BFOLs represented 1.36% (n=383). The ODs (n=187, 48.8%) were the most frequently diagnosed, followed by FD (n=103, 26.9%) and OF (n= 93, 24.3%). Regarding the subtypes, the most common were FlorOD and MFD, both with 101 cases. Women were the majority in the study, with frequency of 82.0% (n=314), as well as non-black patients with (n=226, 59.0%). Among all LFOBs the average age was 38.52 ± 17.54, affecting more the 4th and 5th decades. The most common radiographic features were mixed images: radiopaque and radiolucent (51.7%). The mandible was the most affected region (n=247, 64.5%), except for DF involving more maxilla (n=68, 66.0%). The DF was observed more in men than in women, as well as among younger patients. DOs were more common in women and older patients. The FO was no significant difference between genders. The association between the presence of osteomyelitis and OD was statistically significant (p=0.0001). The swelling was significant in FD and FO (p=0.0001). The frequency of LFOB is similar to that previously reported in the literature in the same country, but differs from data observed in other populations. It is important to consider the correlation of clinical, radiographic and histopathologic for definitive diagnosis of LFOBs. Epidemiological studies can guide the clinician on the patient's profile affected by a disease. Our results are important to assist in the diagnosis and clinical management of benign fibro-osseous lesions

Extra-nodal primary diffuse large B-cell lymphoma of the maxilla. Fine needle aspiration cytology.

A 76-year-old female presented at University hospital of Crete with a large painless mass (d<10 cm) of the left maxilla. The cytologic diagnosis in FNAB smears was of a diffuse large B-cell lymphoma of the maxilla that was confirmed histologically. The fine needle aspiration cytology (FNAC) in conjunction with immunocytochemistry can distinguish between benign and malignant lymphoid infiltrates and support a diagnosis of extra-nodal diffuse large B-cell lymphoma.

Radiographic analysis of ameloblastoma: a retrospective study.

BACKGROUND: Ameloblastoma is benign odontogenic tumor, usually affecting the posterior region of mandible. It is seen in the third to fifth decades of life. Radiographically the lesion is variable in appearance and may be unilocular or multilocular, with well-defined cortical borders in the mandible and ill-defined margins in the maxilla. OBJECTIVE: To analyze cases of ameloblastoma, with emphasis on the radiographic findings. We also review the current literature briefly and discuss the clinical and radiographic findings. MATERIALS AND METHODS: The present hospital-based retrospective study was conducted by reviewing the clinical and radiographic records of ameloblastoma cases from 2009 to 2011, available in the archives of the department. The data of a total of 14 patients were analyzed. RESULTS: We observed that the patients affected with ameloblastoma were in the age-group of 19-68 years. The male: female ratio was 1.3:1. The mandible (78.57%) was more commonly affected than the maxilla (14.28%). Six patients (42.86%) had unilateral involvement and eight cases (57.14%) had bilateral involvement. The multilocular and unilocular types of ameloblastoma were noted in 12 (85.72%) and 2 cases (14.28%), respectively. The soap-bubble (50.00%), spider-web (21.43%), and honeycomb (14.28%) appearances were seen in the multilocular variety. Root resorption of variable degree was distinctly observed in 11 cases (78.57%). CONCLUSION: Radiographs are an important aid for the diagnosis of oral lesions of various types, especially those that involve bone. It is important for the practicing clinicians to know the salient features of ameloblastoma which are peculiar to the local population.

Case of adenomatid odontogenic tumor during pregnancy.

Adenomatoid odontogenic tumor (AOT) is a relatively rare benign tumor, accounting for 2% to 7% of odontogenic tumors. AOT is generally found in the incisor region of the maxilla of young women. It is often misdiagnosed as a dentigerous cyst. We report a case of AOT during pregnancy. The patient was a 21-year-old Philippine woman who complained of a swelling in the right maxillary gingival; the swelling rapidly enlarged during pregnancy. We enucleated the tumor and the impacted canine, with the patient under general anesthesia. Histological examination revealed a pseudoglandular structure with odontogenic appearance. On the basis of these findings and the World health organization (WHO) classification, we diagnosed the tumor as an adenomatoid odontogenic tumor. The tumor cells were identified by an immunohistochemically positive reaction for Bcl-2 and estrogen receptor. The MIB-1 labeling index was less than 1%. There has been no sign of recurrence as of 1.5 years after the operation.

Thirty years clinicopathological study of 60 calcifying cystic odontogenic tumors in Iranian population.

AIM: To reevaluate all cases previously diagnosed as calcifying odontogenic cyst (COC) from the archives of the Department of Oral Pathology at Mashhad Faculty of Dentistry from 1978 to 2008. MATERIALS AND METHODS: Clinical histories and microscopic slides of all cases diagnosed as COC were reviewed and analyzed statistically. The information about patients such as age, sex, clinical features, radiographic view and other necessary data was gathered from biopsy files. RESULTS: The majority of cases occurred in mandible and four cases were peripherally. Thirteen cases were associated with other odontogenic tumors and 11 cases were odontoma type. In two cases, recurrence was reported after surgical treatment which underwent malignant transformation. CONCLUSION: Calcifying cystic odontogenic tumor (CCOT) in Iranian population occurs in younger age group with slightly male predilection and more mandibular, unicystic and simple type. CLINICAL SIGNIFICANCE: It is better to address the noncystic CCOT lesions with their specific terms in order to make histopathological characteristics clear.

[Classification of the formations from blood vessels of maxillofacial region and neck in children].

Multidisciplinary group of investigators basing upon special literature study, analysis of their own observation (1168 cases) including retrospective for 20 years and with the help of several following methods - clinical, roentgenological, pathomorphological,immunohistochemical - picked out from wide group of the so called hemangiomas 3 types of lesions: hyperplasia, malformation, tumour and suggested their clinical biological classification. To each of lesion types characteristic was given.

[Microvascular free flap reconstructive options in patients with different types of maxillectomy defects].

OBJECTIVE: To evaluate the efficacy of the distinct free flaps in reconstruction of different types of maxillectomy defects. METHODS: A retrospective reviews was performed of in 66 consecutive cases of reconstructions with free flaps for maxillary tumor ablation defects from October 1997 to June 2010. There were 43 patients who had recurrences after previous operations and 46 patients had accepted radiation therapy before. According to the classification of Brown's maxilla defect:10 cases were in class I, 13 in class II, 23 in class III and 20 in class IV. The reconstructive free flaps included 26 fibula flaps, 10 radial forearm flaps, 7 latissimus dorsi flaps, 7 rectus abdominis flaps, 7 anterolateral thigh perforator flaps, 5 deep inferior epigastric artery perforators, 2 latissimus dorsi/rib flaps and 2 iliac crest flaps. Postoperative features and functions were assessed in 29 patients. RESULTS: The overall free flap success rate was 93.9% (62/66). Three rectus abdominis flaps and one fibula flap failed. There were 29 patients who received postoperative function assessment. Sixty-two percent of the patients restored to taking regular diets, 24 (82.8%) patients had normal language communication ability, and 25 (86.2%) patients were satisfied with their feature. CONCLUSIONS: Radial forearm flap was recommended to reconstruct the class 1 defect, fibula flap to class 2 or class 3 and preformatted flap to class 4.

Primary intraosseous carcinoma of the anterior maxilla: an unusual case and review of the literature.

Primary intraosseous carcinoma of the head and neck is rare, and lesions isolated to the anterior maxilla are obviously even more rare. The diagnosis of intraosseous carcinoma can be difficult, and it must be based on strict criteria. We report the case of a 40-year-old man who was referred for definitive treatment of a cancerous lesion of the anterior maxilla. The patient's mass had initially been thought to be a benign odontogenic cyst. The patient was treated with surgery and prosthetic rehabilitation. We describe the clinical presentation, pertinent workup including imaging, and the treatment of this case, and we review the literature.

Histopathologic diversity of Gorlin's cyst: a study of four cases and review of literature.

AIM: The purpose of the present article is to discuss four different case reports of the so-called calcifying odontogenic cyst and highlight the histopathological diversity of the same. BACKGROUND: Calcifying odontogenic cyst was first described by Gorlin et al in 1962. Ever since, its identification as a specific odontogenic lesion, controversies and confusions have existed regarding the relationship between cystic lesions and solid tumor masses that share cellular and histomorphologic features. Although several classifications were proposed, dilemma still persists regarding the nature of these lesions as cysts, neoplasms and even malignancies. CONCLUSION AND CLINICAL SIGNIFICANCE: The classifications discussed for the so-called calcifying odontogenic cyst by various authors have only added to further confusion rather than enlightening. Though many authors state that classifications remain only an academic exercise, it definitely has significance in treatment planning. Emphasis should, therefore, be laid on a universally accepted classification.

Notch4 overexpression in ameloblastoma correlates with the solid/multicystic phenotype.

OBJECTIVE: Notch signaling has been implicated in cell fate decisions during odontogenesis and tumorigenesis of some odontogenic neoplasms; however, its role in solid/multicystic (SA), unicystic (UA), and recurrent (RA) ameloblastoma remains unclear. The aim of this study was to determine Notch receptor and ligand expressions in these subtypes and to speculate on their significance. METHODS: Notch receptors (Notch1, 2, 3, 4) and ligands (Jagged1, 2, and Delta1) were examined immunohistochemically in SA (n = 23), UA (n = 22), and RA (n = 19). RESULTS: Notch4 overexpression in SA (n = 19/23; 82.6%) compared with UA (n = 1/22; 4.5%) or RA (n = 10/19; 52.6%) (P < .05) suggests positive correlation between Notch4 signaling and ameloblastomas with a solid/multicystic phenotype. Ligand (Jagged1 and Delta1) underexpression compared with their receptors (Notch1, 3, 4) (P < .05) and nonreactivity for Notch2 and Jagged2 in all 3 subsets suggests that ameloblastoma epithelium belongs to an earlier stage of differentiation (equivalent to inner enamel epithelium of developing tooth germ) before lineage commitment. CONCLUSION: Present findings suggest that Notch signaling molecules may play differing roles in the acquisition of different ameloblastoma phenotypes.

Angiographic classification and sclerotic therapy of maxillofacial cavernous haemangiomas: a report of 204 cases.

A total of 204 patients with maxillofacial cavernous haemangiomas were examined by digital subtraction angiography to investigate the factors affecting therapeutic outcome and to optimize treatment selection. Cavernous haemangiomas were classified as high- or low-drainage based on the nature of the draining veins. Patients were randomized to receive either embolization of the draining veins with absolute ethanol followed by bleomycin A5 hydrochloride intra-tumoural injection, or intra-tumoural injection only. In patients with high-drainage haemangiomas (n = 140), there were significant improvements in the complete cure rate and the overall effective rate in those who had received embolization prior to intra-tumoural injection compared with those who had received intra-tumoural injection only. In patients with low-drainage haemangiomas (n = 64), there were no significant differences between the two treatment groups. It is concluded that embolotherapy of draining veins prior to hydrochloride injection is effective for treating high-drainage cavernous haemangiomas whereas bleomycin A5 hydrochloride injection alone is suitable for treating low-drainage cavernous haemangiomas.

New classification of maxillary ameloblastic carcinoma based on an evidence-based literature review over the last 60 years.

BACKGROUND: The ameloblastic carcinoma is a rare malignant odontogenic tumor which rather occurs in the mandible than in the maxilla. Its rarity and in this context somewhat speculative histopathogenesis may account for diagnostic difficulties. Current classifications do not consider benign histopathological features at the primary and malignant features at the metastatic tumour site. Based on an evidence-based literature review, a recommendation for a novel classification is presented. METHODS: An evidence-based literature review over the last 60 years regarding ameloblastic carcinoma of the maxilla was conducted. RESULTS: An overall of 26 cases were found (mean age: 54.4 (583 years); male to female ratio: 2.7 to 1). In 54% the primary diagnosis was ameloblastic carcinoma, 34.6% revealed pulmonary metastases, however, only in one patient cervical lymph node metastasis could be found. Whereas two cases did not reveal malignant histopathology at the primary, they revealed malignant features at their metastatic sites. Nineteen of 26 patients (73,1%) were controlled during a median follow-up time of 54,3 months (6 to 156 months); 6 patients died of disease after a median time of 62,7 months (7 to 156 months) after initial diagnosis. CONCLUSION: It is of utmost importance to be aware of that ameloblastomas may be capable to degenerate into a "malignant" disease with recurrence and metastasis. In addition to local long-term control, special attention should be paid to potential pulmonary involvement.

Odontogenic tumors in childhood: a retrospective study of 86 treated cases. Importance of a correct histopathologic diagnosis.

The odontogenic tumors are an unusual group of lesions of the jaws derived from embryologic tooth-forming tissues and presenting in a large number of histologic patterns. More common in pediatric age and adolescence than in adult age, the odontogenic tumors can be observed casually or after the appearance of nonspecific symptoms. Because of their slow-growth tendency, usually they do not cause pain. The odontogenic tumors grow in the jaw, through the haversian system, without metastasis but with and high probability of relapse. A retrospective study of 86 cases treated between 1997 and 2005 is reported. The percent of diagnosed cases that were benign was 98.8%, and just one case of malign neoplasm is reported. The most frequent tumor accounted for in the reported sample was odontoma (39.5%) followed by odontogenic fibroma (12.8%). Ameloblastoma and myxoma showed the same incidence (11.6%). Early diagnosis, together with a correct histologic diagnosis, allows a preservative and effective surgical treatment and is necessary to reduce the risk of relapse.

Odontogenic tumors in Sri Lanka: analysis of 226 cases.

PURPOSE: The purpose of this study was to analyze the relative prevalence of odontogenic tumors in Sri Lanka and compare it with prevalences reported for other countries. PATIENTS AND METHODS: A total of 226 cases of odontogenic tumors reported by the Department of Oral Pathology, Faculty of Dental Sciences, University of Peradeniya between 1996 and 2002, were analyzed. RESULTS: The most prevalent tumor was ameloblastoma (69.8%); odontoma was the fourth most prevalent (4.5%). No gender predilection was seen, but the tumors occurred most frequently in the second to fifth decades of life (77.4%) and most commonly in the mandible (79.6%). CONCLUSIONS: The comparative data with statistically significant differences suggest a geographical difference in the relative prevalence of ameloblastoma among various continents. It is suggested that ameloblastoma most frequently occurs in the mandible (the posterior region) in all Asian, African, and South American countries.

[Benign maxillofacial tumours. Odontogenic tumours]. / Tumoraciones maxilares benignas. tumores odontogénicos.

Regarding the 2004 actualization of the classification of benign odontogenic tumours published by IARC and WHO, we have reviewed our files in order to explain the new parameters established in this actualization. Histologically tree groups can be considered depending on the tissue involved: a) odontogenic epithelium with mature fibrous stroma without odontogenic ectomesenchyme, b) odontogenic epithelium with odontogenic ectomesenchyme, with or without tissue formation, and c) mesenchyme and/or odontogenic ectomesenchyme, with or without odontogenic epithelium. Every tumour appears with clinical features, radiographical and specific epidemiology data to complete the cases, in adittion to recurrences and appropriate surgical resection.

Odontogenic tumours: a retrospective study of 1642 cases in a Chinese population.

A total of 1642 odontogenic tumour cases retrieved from the files of the College of Stomatology, Sichuan University, China were retrospectively analyzed for gender, age, tumour site and relative frequency of various types, and the data compared with that of previous reports. The final diagnosis in each case was based on the WHO 2005 histopathological classification of odontogenic tumours. Of these tumours 1592 (97.0%) were benign and 50 (3.0%) were malignant. Ameloblastoma (40.3%) was the most frequent type, followed by keratocystic odontogenic tumour (35.8%), odontoma (4.7%) and odontogenic myxoma (4.6%). The mean age of the patients was 32.1, with a wide range (3-84 years). The male-female ratio and maxilla-mandible ratio were 1.4:1 and 1:4.0, respectively. Ameloblastoma and keratocystic odontogenic tumours, important indications of extensive surgical procedures, are not considered rare in this Chinese population, whereas odontoma is uncommon.

Ameloblastoma: a surgeon's dilemma.

PURPOSE: To investigate whether there were any significant differences in the mode of presentation, treatment, and outcome of patients presenting with a primary diagnosis of ameloblastoma in Glasgow, Scotland and San Francisco, CA. MATERIALS AND METHODS: All cases of ameloblastoma seen in both institutions between January 1, 1980 and December 31, 1999 were included in this study. Mode of presentation, radiographic appearance, histologic appearance, treatment, and follow-up were recorded. RESULTS: There were no significant differences in the clinical features on presentation (swelling, followed by pain, and altered sensation), the radiographic appearance (unilocular approximately 30% and multilocular 70%), or management with either local treatment (enucleation and/or curettage in just over 50% of cases) or radical treatment (a form of resection in under 50%) in the 50 cases included in this study. Primary care by conservative treatment led to a recurrence in approximately 80% of cases and this included cases of unicystic ameloblastoma. CONCLUSION: The mode of presentation, diagnosis, and management of the ameloblastoma was remarkably similar in Glasgow and San Francisco. The recurrence rate following local enucleation and curettage was unacceptably high, and this included the cases of unicystic ameloblastoma, which should be treated more aggressively than has been recommended in the past.