Atypical cellular neurothekeoma of the oral mucosa: A rare case report and literature review.

Cellular neurothekeoma (CN) is a benign dermal neoplasm that most often affects the head and neck region and rarely occurs in the oral mucosa. We report a rare case of CN with atypical features on the floor of the mouth and summarize the reported cases of oral CN in English-language literature. A 62-year-old woman presented with a 6-month history of a painless mass on the floor of the mouth. Histopathological analysis of the excised specimen revealed a proliferation of neoplastic cells with oval to spindle morphology arranged in a vaguely nested and multinodular architecture separated by scarce hyaline collagen within a predominantly myxoid-rich stroma. The tumor cells were positive for NSE, and CD63 (NKI/C3), and negative for S100 protein, CD34, and SMA. Thus, the final diagnosis was CN. In addition, we summarized all clinicopathological data on oral CNs reported in the English-language literature. Nineteen cases were reviewed. Among them, only one case affected the floor of the mouth of a young girl, in contrast to the present case that occurred in an elderly woman. CN is particularly rare in this location and may be a diagnostic challenge for oral pathologists due to its rarity and morphological similarity with other lesions.

Gingival metastasis of angiosarcoma of the breast as a first manifestation of spreading disease: Case report and review of the literature.

BACKGROUND: Angiosarcoma is an aggressive malignant neoplasm of vascular origin. Oral metastases of angiosarcoma are rare and have a non-specific clinical presentation, thus the diagnosis may be challenging. CASE REPORT: Herein we report a case of a 34-year-old female patient after treatment of a high-grade angiosarcoma of the breast, who presented an asymptomatic bleeding purplish nodule in the maxillary interdental papilla between the first and second premolar. A biopsy was performed, and the histological examination revealed infiltration by malignant neoplasm of epithelioid and fusocellular pattern. Immunohistochemical analysis demonstrated that neoplastic cells were positive for ERG and CD31, and negative for cytokeratins AE1/AE3, confirming the diagnosis of metastatic angiosarcoma. After investigation, multiple metastases were discovered. The patient is under management with chemotherapy and palliative radiotherapy for the bone lesions. CONCLUSION: Metastases should be considered in the differential diagnosis of oral lesions in patients with a previous history of cancer. Due to the morphology of angiosarcomas, the metastatic lesions may resemble benign vascular lesions, therefore, biopsy is mandatory to exclude malignancy.

Gingival cyst of the adult.

The gingival cyst of the adult (GCA) is a rare odontogenic cyst, consisting of 0.3% of all odontogenic cysts. This case report, based on CARE guidelines for case reports, aims to present a case of a 52-year-old female patient with a symptomatic translucent nodule in the upper left anterior gingiva, measuring approximately 6mm. Excisional biopsy was performed, and the histological examination revealed multiple cystic cavities lined by the squamous epithelium of varying thickness with focal areas of nodular thickenings. The presence of clusters of cells with clear cytoplasm within epithelial thickenings was observed. PAS staining was negative in clear cells. The diagnosis of the GCA was established. Despite its rarity, GCA should be considered in the differential diagnosis of gingival lesions. Conservative surgical treatment proved to be effective, with no signs of recurrence.

3D printing technology to produce intraoral stents for head and neck radiotherapy: A scoping review.

INTRODUCTION: Radiotherapy remains one of the main treatments for head and neck cancer; however, it is accompanied by acute and chronic adverse effects. Use of three-dimensional (3D) oral stents to modulate radiation intensity to specific target areas have been developed to minimize these adverse effects. This study aimed to present a scoping review of studies published on 3D printing of oral stents and their clinical applicability. METHODS: MEDLINE/Pubmed, Scopus, Web of Science and CENTRAL Cochrane data bases were searched, studies selected, and data collected by three independent reviewers up to December 2022. The review was conducted based on the Preferred Reporting Items for Systematic Reviews and Meta-analysis-Extension for Scoping Reviews (PRISMA-ScR). RESULTS: The search resulted in 404 studies and 5 articles fulfilled the eligibility criteria and were considered for this review. Three-dimensional printed intraoral stents were produced for 56 patients with indication for radiotherapy. 3D-printed stents were well-tolerated by all tested patients and demonstrated great reproducibility of maxillomandibular relation, required less time for production and lower cost to manufacture. Two studies showed great protection of healthy tissues with 3D-printed stents during radiotherapy. CONCLUSIONS: Three-dimensional printing is promising for production of intraoral stents, however, more studies are needed to improve the technique and further investigate the safety and prevention of oral toxicities from radiotherapy.

Gingival cyst of the adult

ABSTRACT The gingival cyst of the adult (GCA) is a rare odontogenic cyst, consisting of 0.3% of all odontogenic cysts. This case report, based on CARE guidelines for case reports, aims to present a case of a 52-year-old female patient with a symptomatic translucent nodule in the upper left anterior gingiva, measuring approximately 6mm. Excisional biopsy was performed, and the histological examination revealed multiple cystic cavities lined by the squamous epithelium of varying thickness with focal areas of nodular thickenings. The presence of clusters of cells with clear cytoplasm within epithelial thickenings was observed. PAS staining was negative in clear cells. The diagnosis of the GCA was established. Despite its rarity, GCA should be considered in the differential diagnosis of gingival lesions. Conservative surgical treatment proved to be effective, with no signs of recurrence.

A rare case of Ewing sarcoma metastasis to the oral cavity.

Ewing sarcoma in the head and neck is rare, and metastasis from other bones to the mandible accounts for 0.7% of cases. This report presents a case of oral metastasis in a 24-year-old male patient diagnosed with Ewing sarcoma of the femur (p53 gene mutation and EWSR1-ERG fusion). The chief complaint was numbness in the mandible and pain for 1 month and a hardened, ulcerated exophytic lesion in the right retromolar region. Imaging exams revealed an unspecified thinning of the cortical bone of the inferior alveolar canal in the right mandibular ramus, associated with erosion of the alveolar bone. Histopathological analysis confirmed metastasis of Ewing sarcoma. The patient presented an aggressive disease progression and died 1 month after the oral diagnosis. It is important to recognize the signs and symptoms compatible with rare clinical outcomes, leading to an early diagnosis that can improve patients' quality of life and survival.

Mandible metastasis of small cell lung cancer mimicking a residual cyst.

Small cell lung carcinoma (SCLC) is an uncommon tumor characterized by an aggressive behavior with early metastasis, usually to the contralateral lung, liver, brain, and bones. There are only five cases of this particular tumor metastasizing to the oral cavity described in the English literature. We present the case of metastatic SCLC in the mandible with radiographic findings resembling a residual cyst. A 66-year-old man with previous diagnosis and treatment for a SCLC was referred to the Stomatology Department with a history of persistent pain in the mandible 1 year after the inferior right pre-molar tooth extraction. The radiographic exam showed a well-delimited radiolucent area on that extracted tooth's region resembling a residual cyst. Biopsy was performed yielding the diagnosis of metastatic SCLC. The patient was referred to the clinical oncologist for chemotherapy. Although uncommon, this tumor should be included in the differential diagnosis of jawbone lesions, particularly when the patient presents a previous diagnosis of SCLC.

Mandible metastasis of small cell lung cancer mimicking a residual cyst

Small cell lung carcinoma (SCLC) is an uncommon tumor characterized by an aggressive behavior with early metastasis, usually to the contralateral lung, liver, brain, and bones. There are only five cases of this particular tumor metastasizing to the oral cavity described in the English literature. We present the case of metastatic SCLC in the mandible with radiographic findings resembling a residual cyst. A 66-year-old man with previous diagnosis and treatment for a SCLC was referred to the Stomatology Department with a history of persistent pain in the mandible 1 year after the inferior right pre-molar tooth extraction. The radiographic exam showed a well-delimited radiolucent area on that extracted tooth's region resembling a residual cyst. Biopsy was performed yielding the diagnosis of metastatic SCLC. The patient was referred to the clinical oncologist for chemotherapy. Although uncommon, this tumor should be included in the differential diagnosis of jawbone lesions, particularly when the patient presents a previous diagnosis of SCLC.

Amlodipine-induced gingival overgrowth in a child after liver transplant.

Drug-induced gingival overgrowth (GO) has been associated with phenytoin, cyclosporine, and calcium channel blocker therapies. This study reports the case of an 11-year-old girl who was referred for evaluation of GO, which had occurred over the last 6 months. Her medical history included a liver transplant due to biliary atresia 3 years ago, immunosuppressive therapy, and hypertension, which is why she was started on a daily intake of amlodipine. The intraoral examination showed generalized GO, and the treatment consisted of a gingivectomy. Subsequently, amlodipine was replaced with captopril and oral hygiene instructions. There was no recurrence of GO after 28 months of follow-up. Although GO may be related to the chronic use of amlodipine, such an association is uncommon in pediatrics, and the treatment consists of the replacement of medication combined with a surgical approach and plaque control.

Surgical Therapy for Bisphosphonate-Related Osteonecrosis of the Jaw: Six-Year Experience of a Single Institution.

PURPOSE: Bisphosphonates are drugs commonly used to treat osteoporosis, hypercalcemia of malignancy, and bone metastases. In some cases, its administration has been associated with osteonecrosis of the jaws. The management of medication-related osteonecrosis of the jaw (MRONJ) has not been completely elucidated, and its treatment can vary from no or limited surgery to more extensive surgery. The objective of the present study was to evaluate the efficacy of surgical therapy for patients presenting with MRONJ. PATIENTS AND METHODS: A retrospective study was conducted that evaluated all MRONJ cases resulting from bisphosphonate use and treated by surgery from 2006 to 2012. All patients underwent surgery under general anesthesia. RESULTS: A total of 33 patients with 46 MRONJ sites were evaluated. Most of the patients were women, with an age range of 39 to 83 years (mean 65.6 ± 10.6). Complete healing of the MRONJ region was observed in 40 of the 46 sites (87%), with partial improvement (symptom control and reduction of the exposed bone area) observed in 3 sites (6.5%), for a 93.5% clinical benefit rate. Of the remaining regions, 2 showed no significant changes, and 1 presented with a worse aspect compared with the patient's preoperative condition. Such cases were located in the posterior mandible region. The number of applications and type of bisphosphonate did not influence the treatment response. CONCLUSION: The surgical approach to treating MRONJ showed a high rate of clinical control. Therefore, surgery should be considered as a therapy for some cases of this condition.

Head and neck solitary infantile myofibroma: Clinicopathological and immunohistochemical features of a case series.

Infantile myofibroma is a rare mesenchymal benign tumor mostly found in the head and neck region. The aim of this study was to describe a small case series of head and neck solitary infantile myofibroma, emphasizing the importance of the histopathological and immunohistochemical features, and the potential diagnostic challenges. For the study, clinical and imaging data were obtained from the medical records. All cases were histologically reviewed, and immunohistochemical analyses were performed to confirm the diagnosis. Four cases of head and neck solitary infantile myofibroma were identified. All patients were females and presented a mean age of 3 years old (ranging from 2 to 6 years). The site of the tumors were the mandible, right cheek, subcutaneous tissue adjacent to basal cortical of the mandible and upper anterior gingiva. No symptoms, such as pain or paresthesia, were reported. Computerized tomography revealed well-delimited tumors. All tumors were positive for vimentin and alpha-smooth muscle actin. All patients underwent surgical excision and no signs of recurrence were observed after long-term follow-up. In summary, head and neck solitary infantile myofibromas are rare and present excellent prognosis. The correlation between clinical, histopathological and immunohistochemical features are essential for a correct diagnosis.

Osteonecrosis related to once-yearly zoledronic acid treatment in an osteoporotic patient after dental implant.

This paper presents a case of osteonecrosis of the jaw related to zoledronic acid (5 mg) administered once yearly to treat osteoporosis. A 79-year-old woman who has been treated for osteoporosis for 5 years with 5 applications of zoledronic acid was referred for evaluation. The patient had been submitted to dental implant placement and there was no osseointegration. On clinical examination, suppuration and exposed bone on the alveolar ridge were observed. Radiographic examination revealed an osteolytic area and bone sequestration. Both clinical and radiological features were suggestive of osteonecrosis. The treatment consisted of surgery to remove the affected bone completely. The patient is asymptomatic at 9 months after surgery. Dentists and oral surgeons should be alert to the possibility of osteonecrosis related to the use of once-yearly injections of zoledronic acid for the treatment of postmenopausal osteoporosis.

Osteonecrosis Related to Once-Yearly Zoledronic Acid Treatment in an Osteoporotic Patient after Dental Implant

This paper presents a case of osteonecrosis of the jaw related to zoledronic acid (5 mg) administered once yearly to treat osteoporosis. A 79-year-old woman who has been treated for osteoporosis for 5 years with 5 applications of zoledronic acid was referred for evaluation. The patient had been submitted to dental implant placement and there was no osseointegration. On clinical examination, suppuration and exposed bone on the alveolar ridge were observed. Radiographic examination revealed an osteolytic area and bone sequestration. Both clinical and radiological features were suggestive of osteonecrosis. The treatment consisted of surgery to remove the affected bone completely. The patient is asymptomatic at 9 months after surgery. Dentists and oral surgeons should be alert to the possibility of osteonecrosis related to the use of once-yearly injections of zoledronic acid for the treatment of postmenopausal osteoporosis.

O presente estudo teve como objetivo apresentar um caso de osteonecrose dos maxilares associada ao uso de ácido zoledrônico (5 mg) administrado uma vez ao ano para tratar a osteoporose. Uma mulher de 79 anos de idade estava em tratamento de osteoporose por 5 anos com 5 aplicações do ácido zoledrônico foi encaminhada para nossa avaliação. A paciente tinha sido submetida à colocação de implante dental e não houve osseointegração. Ao exame clínico, supuração e osso exposto no rebordo alveolar foram observados. Os exames radiográficos revelaram uma área osteolítica e sequestro ósseo. Ambos os aspectos clínicos e radiográficos eram sugestivos de osteonecrose. O tratamento consistiu de cirurgia para remover todo o osso afetado. A paciente está assintomática há 9 meses (desde a cirurgia). Cirurgiões-dentistas e cirurgiões orais devem estar atentos para a possibilidade de osteonecrose relacionada ao uso de injeções anuais de ácido zoledrônico para tratamento da osteoporose pós-menopausa.

Evaluation of socket healing in patients undergoing bisphosphonate therapy: experience of a single Institution.

OBJECTIVE: To assess the clinical features of exodontias performed in cancer patients who have been receiving intravenous bisphosphonates (BPs). STUDY DESIGN: This is a retrospective cohort study using a sample of 20 patients receiving BPs who had 62 teeth extracted. An univariate analysis was applied to calculate socket healing time (HT), comparing among exodontias performed according to cause, such as periodontal disease or caries, type of BP, and use of corticosteroid. In order to analyze the influence of each variable on HT, multiple statistical analyses were performed through logistic multiple regression. RESULTS: From the 62 tooth extractions performed, 5 exodontias had evolved to 4 sites of bisphosphonate-related osteonecrosis of the jaws (BOJ). Of another 57 exodontias without development of BOJ, HT was significantly better for tooth extraction performed in patients receiving corticosteroid (p= .01), for tooth extracted due to caries (p= .04), and for extractions under pamidronate (p= .03). Sockets after exodontias due to periodontal diseases had OR= 5.22 (95% CI 1.73-133.66, p=0.01) for delayed HT, exodontias performed under corticosteroid use had OR=0.04 (95% CI 0.01-0.40, p<0.001), and exodontias performed under zoledronate had OR=0.31 (95% CI 0.08-1.25, p=0.10). CONCLUSIONS: Exodontias performed in patients under BP therapy had a low rate of BOJ occurrence. Zoledronate and periodontal diseases influence delayed socket healing. Adjuvant antibiotics could be relevant procedures aimed at reducing the risk of BOJ development.

Jaw osteonecrosis in patients receiving bisphosphonate therapy: results from a single institution

A growing number of reported cases of jaw osteonecrosis in patients receiving bisphosphonate have been published in the last several years. The clinical features of this condition include pain, paresthesia, bone exposure and fistula. Risk factors have been recognized and classified as local and/or systemic. Objective: The aim of this study was to demonstrate the clinical data of the patients with osteonecrosis assisted at a single institution. Patients and Methods: A total of 42 patients presenting 49 areas of jaw osteonecrosis were evaluated. Medical records were analyzed in order to collect information on underlying disease, bisphosphonate information, clinical features related to bisphosphonate-induced osteonecrosis of the jaw, as well as precipitating events related to its occurrence. Results: Most patients were female (71%) and the mean age was 64.7 years old. Breast cancer was the most frequent underlying disease (40.5%) followed by multiple myeloma, prostate cancer, lung and osteoporosis. In addition, the average use of bisphosphonate was 36.8 months and most patients had received zoledronic acid. The posterior region of the mandible was the main affected site. Among the possible triggering factors, exodontias was associated with 73.8% of the cases. The treatment modalities consisted of surgical, local irrigation with clorexidin and antibiotics and a majority of cases presented complete or partial remission. Conclusion: Most cases of jaw osteonecrosis were related to tooth extraction and surgical interventions showed a good control of the osteonecrosis cases.

Avaliação da resposta ao tratamento cirúrgico radical em pacientes portadores de osteonecrose dos maxilares associada ao uso de bisfosfonatos / Evaluation of response of radical surgical treatment in patients with bisphosphonate associated osteonecrosis of the jaws

Bisfosfonatos são drogas comumente prescritas para o controle de osteoporose, tratamento de hipercalcemia maligna e controle de metástases ósseas. Entretanto, recentemente alguns pacientes que receberam esta terapia têm desenvolvido osteonecrose nos ossos maxilares. O tratamento dessa alteração ainda não está elucidado e alguns casos têm evoluído de forma insatisfatória. Os tratamentos descritos variam de conservadores, como antibioticoterapia, cuidados locais e debridamentos superficiais a cirurgias radicais com remoção de todo osso afetado. Há, no entanto, poucos estudos relacionados ao sucesso do tratamento cirúrgico da osteonecrose. Neste sentido, o objetivo deste estudo foi avaliar a resposta ao tratamento cirúrgico radical, em pacientes portadores de osteonecrose dos maxilares associada ao uso de bisfosfonatos e determinar possíveis fatores que possam influenciar a resposta do tratamento. Foi realizado um estudo de coorte retrospectivo que analisou os pacientes atendidos no Departamento de Estomatologia do Hospital A.C. Camargo no período de 2004 à 2010. A amostra constitui-se de 33 pacientes, os quais apresentaram 46 áreas de osteonecrose. Os resultados obtidos mostraram uma taxa de cicatrização completa de 87%. Além disto, outros 7% apresentaram melhora parcial, ou seja, controle dos sintomas e redução da área de exposição óssea, traduzindo em 94% de benefício clínico utilizando esta terapia. Entre as regiões restantes, 4% não apresentaram modificações significantes e 2% apresentaram piora em relação ao aspecto pré-operatório, sendo que estes casos localizavam-se na região posterior da mandíbula. Observamos que o número de aplicações e o tipo de Bisfosfonato não influenciaram a resposta ao tratamento cirúrgico nesta amostra. Concluímos que o tratamento cirúrgico da osteonecrose por bisfosfonatos é efetivo, apresentando índice elevado de cicatrização completa, contudo a região posterior da mandíbula pode apresentar pior resposta cicatricial.

Bisphosphonates are drugs commonly used for treating osteoporosis, hypercalcemia of malignancy and bone metastases. However, the administration of bisphosphonates has been associated with osteonecrosis of the jaws. The treatment of this disorder has not been elucidated and some cases shown unsatisfactory results. The treatments described vary from conservative such as antibiotics, local wound care and superficial debridements to radical surgery with removal of all affected bone. However, few studies showed the success of surgical treatment of osteonecrosis. The objective of this study was to evaluate the response of radical surgical treatment in patients with bisphosphonate associated osteonecrosis of the jaw and determine possible factors that might influence the treatment response. It was conducted a retrospective analysis that evaluated patients treated at Stomatology Department of AC Camargo Hospital, from 2004 to 2010. The study included 33 patients, which showed 46 areas of osteonecrosis. Complete healing rate was 87% and partial improvement (symptom control and reduction of area of exposed bone) was 7%, resulting in 94% of clinical benefit using this therapy. Among the remaning regions, 4% showed no significant changes and 2% worsened compared to preoperative aspect, these cases were located in the posterior mandible region. The number of applications and type of bisphosphonate did not influence the response of surgical treatment in this study. Our data demonstrate that surgical treatment of bisphosphonate associated osteonecrosis of the jaw is effective with high rate of complete healing; however, the posterior mandible region presents worst response.