Giant Mandibular Ameloblastoma with Rare Hypercalcemia: A Case Report and Literature Review.

Ameloblastoma is the most common benign odontogenic tumor with local invasion and high recurrence, which generally occurs in the jaw bones. Hypercalcemia is a common paraneoplastic syndrome that is commonly observed in patients with malignancies but rarely encountered in patients with benign tumors. Thus far, not many cases of ameloblastoma with hypercalcemia have been reported, and the pathogenic mechanism has not been studied in depth. This paper presents a case report of a 26-year-old male diagnosed with giant ameloblastoma of the mandible, accompanied by rare hypercalcemia. Additionally, a review of the relevant literature is conducted. This patient initially underwent marsupialization, yet this treatment was not effective, which indicated that the selection of the appropriate operation is of prime importance for improving the prognosis of patients with ameloblastoma. The tumor not only failed to shrink but gradually increased in size, accompanied by multiple complications including hypercalcemia, renal dysfunction, anemia, and cachexia. Due to the contradiction between the necessity of tumor resection and the patient's poor systemic condition, we implemented a multi-disciplinary team (MDT) meeting to better evaluate this patient's condition and design an individualized treatment strategy. The patient subsequently received a variety of interventions to improve the general conditions until he could tolerate surgery, and finally underwent the successful resection of giant ameloblastoma and reconstruction with vascularized fibular flap. No tumor recurrence or distance metastasis was observed during 5 years of follow-up. Additionally, the absence of hypercalcemia recurrence was also noted.

Simultaneous Management of Condylar Osteochondroma and Secondary Malformation With a New Intraoral Approach.

PURPOSE: The present study aimed to investigate the clinical efficacy of simultaneous management of condylar osteochondroma and its secondary dentofacial deformities using an intraoral surgical approach. METHODS: Six patients with condylar osteochondroma were treated with intraoral vertical ramus osteotomies and condylar resection. The free rising branch was used for reconstructing the temporomandibular joint. The simultaneous orthognathic surgery and plastic surgery were performed sequentially to correct the secondary dentofacial deformities. The indexes of aesthetic symmetry, occlusion relationship, temporomandibular joint function, condylar height, and volume change were assessed in the subsequential follow up. RESULTS: The mean follow up period was 31 months. All patients had no tumor recurrence. The ipsilateral joint function, occlusal relationship, and facial symmetry were satisfied. The ipsilateral condylar reconstruction had no obvious bone resorption and the ramus height was maintained well. Postoperative assessment showed the preoperative design was accurately fulfilled. CONCLUSIONS: The simultaneous condylar osteochondroma resection and temporomandibular joint reconstruction using intraoral approach avoids extraoral scars and correct facial asymmetry without compromising the long-term joint function and occlusal relationship.

Prolonged Hypophosphatemia and Intensive Care After Curative Surgery of Tumor Induced Osteomalacia: A Case Report.

Introduction: Rare FGF23-producing mesenchymal tumors lead to paraneoplastic tumor-induced osteomalacia (TIO) presenting with phosphate wasting, hypophosphatemia, chronic hypomineralization of the bone, fragility fractures and muscle weakness. Diagnosis of TIO requires exclusion of other etiologies and careful search for a mesenchymal tumor that often is very small and can appear anywhere in the body. Surgical removal of the tumor is the only definitive treatment of TIO. Surgical complications due to chronic hypophosphatemia are not well recognized. Case Description: The current case describes severe fragility fractures in a 58-year-old woman, who lost her ability to walk and was bedridden for two years. First, the initial diagnostic laboratory work-up did not include serum phosphorus measurements, second, the suspicion of adverse effects of pioglitazone as an underlying cause delayed correct diagnosis for at least two years. After biochemical discovery of hyperphosphaturic hypophosphatemia at a tertiary referral centre, a FGF23-producing tumor of the mandible was discovered on physical examination, and then surgically removed. Postoperatively, severe hypophosphatemia and muscle weakness prolonged the need for ventilation support, intensive care and phosphate supplementation. After two years of rehabilitation, the patient was able to walk short distances. The tumor has not recurred, and serum phosphate concentration has remained within normal limits during 3.5 years of follow-up. Conclusions: The case report illustrates knowledge gaps in the diagnostic work-up of rare causes of low bone mass and fragility fractures. Compared to other low phosphate conditions, surgical recovery from TIO-induced hypophosphatemia warrants special attention. Increased alkaline phosphatase concentration may indicate impaired postsurgical recovery due to prolonged hypophosphatemia, underlining the need for proactive perioperative correction of hypophosphatemia.

Transfacial transmandibular approach to the masticator space: Excision of odontogenic myxoma arising from the mandibular condyle.

Odontogenic myxomas are an uncommon benign odontogenic tumor that can present with a wide variety of symptomatology depending on location and potentially be locally destructive. The present case describes a 66-year-old female who presented with left lower facial paresthesia, left aural fullness and hearing loss. She was found to have an odontogenic myxoma that involved the condylar head and extended into the masticator space. In this report we detail our surgical approach utilizing a preauricular transfacial transmandibular approach to the masticator space. In addition, we will discuss various approaches to the masticator space and infratemporal fossa along with considerations on how to manage facial nerve paralysis, facial contour deformities, and post-operative rehabilitation for permanent unilateral condylar head disarticulation.

Ameloblastic Fibroodontoma of Mandible Causing Tumor Induced Osteomalacia: A Case Report with Review of 88 Phosphaturic Oral Neoplasms.

Tumor induced osteomalacia (TIO) is a rare paraneoplastic syndrome associated with tumors secreting fibroblast growth factor 23, which induces osteomalacia. Microscopically, these tumors most commonly show benign phosphaturic mesenchymal tumors. We report the first case of phosphaturic ameloblastic fibro-odontoma (AFO) manifesting as osteomalacia. Our index patient was a 33-year-old male who was diagnosed with TIO and AFO in the mandible was identified as the cause. Our case is unique as AFO is considered as a hamartoma. To the best of our knowledge, there is no hamartoma reported till date causing phosphaturic osteomalacia. As AFO demonstrates mixed epithelial and mesenchymal origin, we propose a new histopathological subtype of TIO-"phosphaturic tumor of mixed epithelial and mesenchymal origin". A review of literature focused on TIO caused by oral lesions revealed 88 oral neoplasms which matched our search criteria. Due to the rarity and unpredictable behavior of TIOs, a high index of suspicion, a broad diagnostic approach, detailed history and multidisciplinary investigations are crucial for establishing the definitive diagnosis and proper treatment recommendations.

Central ossifying fibroma of mandible.

Ossifying fibroma (OF) is a rare, benign, fibro-osseous lesion of the jawbone characterised by replacement of the normal bone with fibrous tissue. The fibrous tissue shows varying amount of calcified structures resembling bone and/or cementum. The central variant of OF is rare, and shows predilection for mandible among the jawbone. Although it is classified as fibro-osseous lesion, it clinically behaves as a benign tumour and can grow to large size, causing bony swelling and facial asymmetry. This paper reports a case of large central OF of mandible in a 40-year-old male patient. The lesion was treated by segmental resection of mandible. Reconstruction of the surgical defect was done using avascular fibula bone graft. Role of three-dimensional printing of jaw and its benefits in surgical planning and reconstruction are also highlighted.

Compound odontoma causing impaction of primary tooth in a 4-year-old child: case report / Impacção de dente decíduo por presença de odontoma composto em criança de 4 anos: relato de caso

Objetivo: relatar um caso raro de impacção de um incisivo decíduo inferior pela presença de odontoma composto, bem como descrever a sua abordagem clínica. Relato de caso: paciente do sexo masculino, com 4 anos de idade, apresentava ausência do incisivo lateral decíduo inferior esquerdo. O exame radiográfico mostrou impacção do incisivo não erupcionado próximo a estruturas radiopacas sugestivas de odontoma composto. O paciente foi acompanhado por dois anos, momento em que se realizou abordagem cirúrgica do caso. Após a cirurgia, a hipótese de diagnóstico de odontoma composto foi confirmada e com o acompanhamento ocorreu a erupção dos incisivos centrais permanentes inferiores. O paciente foi encaminhado para tratamento ortodôntico. Considerações finais: esse relato de caso aborda um caso raro de odontoma composto associado à não erupção de dente decíduo, uma vez que odontomas costumam ser detectados preferencialmente na segunda década de vida do paciente, sendo associados à impacção de dentes permanentes. Além disso, apresenta um protocolo de abordagem clínica para esses casos quando diagnosticados em idade precoce no paciente infantil.(AU)

Objective: to report a rare case of impaction of a primary mandibular incisor due to the presence of a compound odontoma and describe its clinical management. Case report: a 4-year-old boy presented with a "missing" primary left mandibular lateral incisor. Radiographs showed impaction of the unerupted incisor by adjacent radiopaque structures consistent with a compound odontoma. The patient was recalled periodically for 2 years, at which time surgical excision was performed. The diagnosis of compound odontoma was confirmed histologically, and the permanent mandibular central incisors erupted uneventfully; the patient was referred for orthodontic treatment. Final considerations: this case report describes an unusual case of compound odontoma associated with an unerupted deciduous tooth; odontomas are rare in this age range, occurring predominantly in the second decade of life and in association with impaction of permanent teeth. We also propose a protocol for clinical management of such early-onset cases.(AU)

Cisto ósseo traumático associado a odontoma composto: relato de um caso incomum / Aneurismal bone cyst with a compound odontoma: unusual case report

Introdução: o cisto ósseo traumático (COT) é um pseudocisto que se apresenta assintomático e é descoberto frequentemente em exames de rotina. Outra lesão também presente nos maxilares é o odontoma, sendo dividido em dois subtipos, o composto e o complexo; os odontomas são geralmente descobertos como um achado acidental, visto que não apresentam sintomatologia. Objetivo: relatar um caso incomum de um COT, associado à odontoma composto (OC). Relato de caso: paciente do gênero masculino, 16 anos de idade, compareceu à clínica escola de odontologia da Universidade Federal de Campina Grande (UFCG), campus Patos, PB, referenciado pelo cirurgião-dentista após solicitar exame radiográfico para tratamento ortodôntico e observar lesão radiolúcida em região anterior da mandíbula. Durante a anamnese, o paciente não relatou nenhuma alteração sistêmica ou doença de base, mas relatou trauma de infância na região acometida. No exame clínico intraoral, não foi observado nenhum aumento de volume na região. Realizou-se palpação na região, não havendo relato de dor. Ao analisar a radiografia panorâmica, observou-se a presença de pequenas estruturas calcificadas com radiopacidade semelhante às estruturas dentárias, delimitada por uma linha radiolúcida, sugestiva de OC. Para melhor localização, delimitação, relação com estruturas anatômicas e planejamento cirúrgico da lesão, foi solicitado um exame de tomografia computadorizada de feixe cônico (TCFC). Considerações finais: com base nos achados clínicos e radiográficos, optou-se por abordagem cirúrgica da lesão cística e enucleação do OC, sob anestesia local. No pós-operatório de um ano, o paciente evoluiu satisfatoriamente sem queixas clínicas.(AU)

Introduction: traumatic bone cyst (TBC) is a pseudocyst that usually presents asymptomatically and is found frequently in routine exams. Another lesion also present in the jaws is odontoma. The odontoma is divided into two subtypes, the compound and the complex; odontomas are usually discovered as an accidental finding, since they do not present symptomatology. Objective: the present article aims to report an unusual case of a TBC associated with a composite odontoma. Case report: a 16-year-old male patient attended the Clinic School of Dentistry of the Universidade Federal de Campina Grande (UFCG), Patos-PB campus, referenced by the dentist after identify radiolucent lesion in the anterior region of the mandible on radiographic examination for orthodontic treatment. During the anamnesis, the patient did not report any systemic alteration or underlying disease, but reported trauma in the region affected in childhood. The intra oral clinical examination, was not observed any increase in volume in the region. Palpation was performed in the region, and there was no report of pain. When analyzing panoramic radiography the presence of small calcified structures with radiopacity similar to dental structures was observed, delimited by a radiolucent line, suggestive of compound odontoma. To better location, delimitation, compared with anatomy and surgical planning of the injury, it was requested an cone beam computed tomography (CBCT). Final considerations: based on the clinical and radiographic findings, we opted for a surgical approach to cystic lesion and enucleation of composite odontoma, under local anesthesia. In the one-year postoperative period, the patient progresses satisfactorily without clinical complaints.(AU)

Evaluation of dentofacial asymmetry caused by odontomas using panoramic radiographs.

AIMS: To evaluate the presence of dentofacial asymmetry in patients with odontoma by panoramic radiography. METHODS: Panoramic images with odontoma were selected among all panoramic radiographs (3058 patients). Maxillary odontoma was detected in 27 patients while mandibular odontoma was detected in 25 patients. In addition, 30 patients with similar age and gender characteristics were selected as the control group. Skeletal angular, skeletal linear and dental measurements were performed on panoramic radiographs. The odontoma region and the opposite side of the odontoma of the individuals were examined. The dentofacial asymmetry of the odontoma groups was compared with the control group. Paired t-test was used to determine dentofacial asymmetry on the right and left side of the patients with odontoma. The ANOVA test was used for testing the differences among groups. RESULTS: As a result of study, no significant difference was found between the region of the odontoma and the symmetrical region in the maxilla and mandibula (P > 0.05). In the control group, a statistically significant difference was found in the angle between the mandibular canal and the mental foramen, lower incisor size, PFH/CutCat(°), and Co-Mc-Me(°) measurements (P < 0.05). In the maxillary and mandibular odontoma groups, a statistically significant difference was found in the angle between the mandibular canal and the menton, CH (mm), RH (mm), and CrH (mm) in the comparison of the odontoma and the control group (P < 0.05). CONCLUSIONS: No difference was found between the right and left sides of the jaws related with the asymmetry of the maxilla and mandible.

Fístula linfática cervical: complicaciones y manejo / Cervical chyle leak: complications and management

La fístula linfática cervical se produce por daño iatrogénico del conducto torácico en cirugías de cabeza y cuello. Ante la sospecha de una fístula linfática se debe iniciar rápidamente un manejo nutricional y seguimiento estrecho, ya que pueden tener consecuencias graves como inmunosupresión, compromiso metabólico y necrosis cutánea con exposición e incluso ruptura carotídea. A continuación se presenta el caso de un paciente con fístula linfática cervical que precisó reintervención quirúrgica para lograr el cese de la fístula

Chyle leak formation is produced by iatrogenic damage to the thoracic duct in head and neck surgeries. Given the suspicion of a cervical chyle leak, nutritional management and close follow-up should be initiated quickly, since they can have serious consequences such as immunosuppression, metabolic compromise and skin necrosis with exposure or even carotid rupture. We report the case of a patient with cervical chyle leak that required surgical reexploration to reach the chyle leak closure

Hybrid ameloblastoma: a mystified histopathological entity.

Ameloblastoma a benign neoplasm of the maxillofacial region has been divided into various histopathological types by WHO. A more complex and confusing type includes hybrid type, which as the name suggest include more than two variants mostly histopathological. Various authors have reported cases of this type but the exact histopathological features are still unclear and each case that is being reported add to the literature, which further strengthens its histopathological feature. Also, this is a lesion whose clinical and radiographical features are similar to all the variants and a definitive diagnosis is achieved by histopathology only. Here, we present a case of hybrid ameloblastoma with striking and unique histopathological features.

Tumor-induced hypophosphatemic osteomalacia caused by a mesenchymal tumor of the mandible managed by a segmental mandibulectomy and microvascular reconstruction with a free fibula flap.

BACKGROUND: Tumor-induced osteomalacia is a rare paraneoplastic syndrome in which patients develop hypophosphatemia and osteomalacia. METHODS AND RESULTS: Here, we report a unique case of a 42-year-old man who presented to our institution with a 1-year history of pain in his ribs, hips, lower back, and feet. Radiologic examination revealed a decrease in bone density and multiple insufficiency fractures. Laboratory evaluation revealed hypophosphatemia, low serum 1,25 dihydroxy vitamin D3 , and elevated fibroblast growth factor 23 (FGF23). A positron emission tomography/CT scan showed increased uptake in the right mandibular third molar region. Panoramic radiography and CT scanning showed a lytic expansile bone lesion. A mandibular bone biopsy revealed a mixed connective tissue tumor. A right segmental mandibulectomy was performed, followed by microvascular reconstruction. The resection was confirmed by normalization of serum phosphate and FGF23. CONCLUSION: Successful management of this condition was achieved, with complete surgical resection of the tumor and reconstructive surgery.

RBM10 truncation in astroblastoma in a patient with history of mandibular ameloblastoma: A case report.

Astroblastoma is a rare glial neoplasm composed of cells that have broad processes oriented perpendicular to central vessels and often demonstrate vascular sclerosis. The WHO 2016 classification does not specify a grading system for astroblastoma, and categorizes them as well-differentiated or malignant. These broad classification rubrics, however, do not accurately predict clinical outcome. Genetic profiling of astroblastoma has therefore been of particular interest in the recent years. These efforts, although in small number, have revealed heterogeneous molecular findings that may explain astroblastoma's unpredictable clinical outcome. Here, we report a case of recurrent astroblastoma in a 23-year-old female with a unique molecular characteristic. Our patient's tumor harbored an RNA-binding motif 10 (RBM10) truncation. RBM10 codes for a widely expressed RNA binding protein, and its mutation has been described in a variety of solid cancers. RBM10 is thought to be involved in stabilization of pro-apoptotic proteins in breast cancer, and its reduced protein expression is associated with advanced stages of lung adenocarcinoma. To our knowledge, this is the first report of astroblastoma harboring RBM10 truncation. Interestingly, our patient also has a history of mandibular ameloblastoma, but the link between these two rare tumors is unclear.

Clinical Study of Reconstruction Plates Used in the Surgery for Mandibular Discontinuity Defect.

BACKGROUND/AIM: Postresective mandibular reconstruction is common in cases of oral and mandibular tumors. However, complications such as plate fracture and/or plate exposure can occur. The purpose of this study was to analyze complications and survival of reconstructive plates used to correct mandibular defects caused by oral cancer. PATIENTS AND METHODS: Clinical and radiological data from 34 patients were analyzed. Only discontinuous mandibular defect cases were included in this study. All cases were classified using the Hashikawa's CAT and Eichner's classification methods. Then, we determined whether these classifications and clinical treatment methods were significantly related to complications. RESULTS: Complications after mandibular reconstruction occurred in 10 of 34 patients, specifically, two plate fractures, one screw fracture, and seven plate exposures occurred. The plate fractures occurred 5 and 6 months after operation, and the screw fracture occurred 39 months after operation. Using the Hashikawa's CAT classification, the two cases of plate fracture were one of AT type and the other of T type, and the screw fracture was AT type. Using Eichner's classification, all three cases of plate and screw fractures were B2 type. CONCLUSION: We suggest that plate and screw fractures were caused by the type of mandibular defect and bite force.

Jaw bones regeneration using mesenchymal stem cells. A single-center experience.

PURPOSE: Mesenchymal stem cells (MSC), which are multipotent stromal cells, are considered to be a promising resource in tissue engineering and tissue regeneration. MSCs have been used to generate new maxillary bone with clinically successful results. The aim of this study was to determine the role of MSC in bone regeneration procedures in patients with benign maxillary lesions. METHODS: A study was conducted on five patients treated for maxillary bone defects resulting from biopsy of benign lesions at the University Hospital of Magna Græcia, Catanzaro, Italy from January 2015 to October 2016. MSC from autologous bone marrow were used for bone regeneration. The bone mineral density was compared, using the Hounsfield scale, before and after treatment. Follow-up was monthly for six months, and the patients underwent a computed tomography scan of the maxilla at 6 months. RESULTS: Five patients, who underwent biopsy of osteolytic odontogenic benign tumors, were included in the study. There were no intraoperative or postoperative complications. The mean volume of the newly formed bone was 2.44cm3 (range 2,0-3,1) and the mean bone density was 1137 Hounsfield Units (range 898-1355). CONCLUSIONS: Bone regeneration with MSC from autologous bone marrow appears to be a valid treatment option for maxillary bone defects. KEY WORDS: Bone regeneration, Mesenchymal stem cells, BM-MSC, Upper jaw, Mandible.

Giant Peripheral Temporomandibular Osteoma With Immediate Reconstruction of Mandible.

Osteoma is a benign osteogenic tumor, which is characterized the slow-growing proliferation of compact or cancellous bone. The etiology of the lesion is not well established. Among the hypotheses, the following are related: a traumatic factor, infectious processes, or true neoplasm due to the origin of cartilaginous cells. It can present at any age, usually in young adult individuals, with equal prevalence in both sexes. The clinical characteristics of the peripheral osteoma are represented by slow asymptomatic growth, which leads to swelling and, thus, facial asymmetry. When it is related to the mandibular condyle, this growth can cause functional impairment, such as malocclusion, temporomandibular joint dysfunction, and even limited mouth-opening owing to ankylosis.

Identification of Human Bocavirus type 4 in a child asymptomatic for respiratory tract infection and acute gastroenteritis - Goiânia, Goiás, Brazil

Abstract Human Bocavirus (HBoV) has been identified from feces and respiratory samples from cases of both acute gastroenteritis and respiratory illness as well as in asymptomatic individuals. The aim of this study was to detect and characterize HBoV from fecal samples collected from hospitalized children aged less than five years old with no symptoms of respiratory tract infection (RTI) or acute gastroenteritis (AGE). The study involved 119 children and one fecal sample was collected from each participant between 2014 and 2015. HBoV was detected using Nested-PCR, and the viral type identified by genomic sequencing. HBoV-4 was identified from one sample obtained from a hospitalized child with soft tissue tumor of the submandibular region. This is the first report of HBoV-4 identification in Brazil, but we consider that this type may be circulating in the country similar to the other types and new investigations are necessary.

Identification of Human Bocavirus type 4 in a child asymptomatic for respiratory tract infection and acute gastroenteritis - Goiânia, Goiás, Brazil.

Human Bocavirus (HBoV) has been identified from feces and respiratory samples from cases of both acute gastroenteritis and respiratory illness as well as in asymptomatic individuals. The aim of this study was to detect and characterize HBoV from fecal samples collected from hospitalized children aged less than five years old with no symptoms of respiratory tract infection (RTI) or acute gastroenteritis (AGE). The study involved 119 children and one fecal sample was collected from each participant between 2014 and 2015. HBoV was detected using Nested-PCR, and the viral type identified by genomic sequencing. HBoV-4 was identified from one sample obtained from a hospitalized child with soft tissue tumor of the submandibular region. This is the first report of HBoV-4 identification in Brazil, but we consider that this type may be circulating in the country similar to the other types and new investigations are necessary.