Mandibular metastases in neuroblastoma: Outcomes and dental sequelae.

BACKGROUND: Although metastatic involvement of bony sites including cranial bones is common in neuroblastoma (NB), mandibular metastases (MM) are uncommon, and specific outcomes have not been reported upon in the modern therapeutic era. METHODS: In this retrospective study, medical records on patients with MM from NB were reviewed. Statistical analysis was performed using the Kaplan-Meier method. RESULTS: Of 29 patients, nine (31%) had MM at diagnosis, whereas in 20 (69%) MM were first detected at NB relapse at a median time of 26 (6-89) months from diagnosis. Median maximal diameter of lesions was 3 (range 0.8-4.9) cm. MM were unilateral in 83% of patients, with ascending ramus (55%) and mandibular body (38%) being the two most common sites. All patients received systemic chemotherapy, and 26 (93%) patients received radiotherapy to MM. At a median follow-up of 37.3 (24.2-219.5) months, eight of nine patients with MM at diagnosis did not experience mandibular progressive disease. Eighteen of 20 patients with MM at relapse received therapeutic radiotherapy; objective responses were noted in 78%. Seventy-two percent (5/18) had not experienced relapse within the radiation field at a median of 12 (2-276) months postradiotherapy. Dental findings at follow-up after completion of NB therapy included hypodontia, hypocalcification of enamel, and trismus. Median 3-year overall survival in patients with relapsed MM was 51 ± 12% months from relapse. CONCLUSION: MM when detected at diagnosis is associated with a prognosis similar to that for other skeletal metastases of NB. Radiotherapy is effective for control of MM detected both at diagnosis and relapse. Significant dental abnormalities posttherapy warrant regular dental evaluations and appropriate intervention.

Whole-Genome and Segmental Homozygosity Confirm Errors in Meiosis as Etiology of Struma Ovarii.

Strumae ovarii are neoplasms composed of normal-appearing thyroid tissue that occur within the ovary and rarely spread to extraovarian sites. A unique case of struma ovarii with widespread dissemination detected 48 years after removal of a pelvic dermoid provided the opportunity to reexamine the molecular nature of this form of neoplasm. One tumor, from the heart, consisting of benign thyroid tissue was found to have whole-genome homozygosity. Another tumor from the right mandible composed of malignant-appearing thyroid tissue showed whole-genome homozygosity and a deletion of 7p, presumably the second hit that transformed it into a cancerous tumor. Specimens from 2 other cases of extraovarian struma confined to the abdomen and 8 of 9 cases of intraovarian struma showed genome-wide segmental homozygosity. These findings confirm errors in meiosis as the origin of struma ovarii. The histological and molecular findings further demonstrate that even when outside the ovary, strumae ovarii can behave nonaggressively until they receive a second hit, thereafter behaving like cancer.

Metastatic Prostate Adenocarcinoma of the Mandible Diagnosed With Oral Manifestations.

Metastasis from the prostate gland to the mandible is rarely encountered and commonly present with non-specific features like unexplained pain, swelling, and numb chin syndrome. Here we present a case with metastatic prostate adenocarcinoma detected secondary to oral manifestations. Patients present with unexplained facial pain and numbness should alert clinicians to the presence of malignant disease, and appropriate hematological, radiological and or histological investigations should be performed. Thereby, clinicians can prevent the overlook of the first signs of metastasis, accelerate the early diagnosis and positively orientate the prognosis of the disease, especially in a patient without known malignancy.

Medulloblastoma With Metastasis to the Jaw in a Child With Nevoid Basal Cell Carcinoma Syndrome: A Case Report.

Medulloblastoma is the most common childhood malignant brain tumor and is considered a minor criterion in the diagnosis of nevoid basal cell carcinoma syndrome (NBCCS). Metastasis of this primitive neuroectodermal tumor is most commonly neuraxial; however, extra-neuraxial metastases have been reported. Extra-neuraxial metastasis to the jaws is a rare occurrence, with only 8 cases previously reported in the literature. The present report documents a unique case of metastatic medulloblastoma to the mandible in a patient with a previous diagnosis of NBCCS.

[Concurrent nodal and extranodal lymphoma misdiagnosed as metastatic gingival carcinoma: A case report].

Early diagnosis of malignant lymphoma is often difficult since the clinical manifestation of the lymphoma occurred in the maxillofacial region is very similar to that of the squamous cell carcinoma. When the pathological diagnosis is not clear, the surgeon is easy to misdiagnose and lead to mistreatment. A patient visited the Affiliated Haikou Hospital of Xiangya School of Medicine, Central South Universily for gingival mass with an ipsilateral submaxillary enlargement. The clinical manifestation and preoperative MRI are very prone to squamous cell carcinoma with metastasis, so we did not take a preoperative pathological examination. The gingival mass was surgical removed firstly, but frozen pathological result showed that it was malignant small round cell tumor. Since the patient was diagnosed as high degree malignant of small round cell tumor and the submandibular region have been significantly metastasized, so we carried out the combined radical dissection of gingival, mandible and neck surgery. The postoperative pathological report was malignant lymphoma, suggesting that the patient was a case of misdiagnosis and mistreatment. This article draws lessons from misdiagnosis and provided experience for seeking improvement measures.

Metastatic retinoblastoma of the parotid and submandibular glands: a rare case report.

BACKGROUND: Retinoblastoma is the most common intraocular malignancy occurring in children. It can metastasize to the regional lymph nodes, central nervous system and distant organs usually the bones and bone marrow and very rarely to the soft tissue. Here, we report a case of unilateral retinoblastoma in a 4-year-old girl accompanied by a large metastasis of the parotid and submandibular glands that developed about 6 months previously and gradually increased in size 5 months after enucleation of the left eye. CASE PRESENTATION: A 4-year-old girl with a history of unilateral retinoblastoma presented with a large, painful and worsening mass (about 20 × 23 cm) of the left side of the neck. Following surgery, the orbital tumour was completely resected, and the large tumour invasion range in the left side of the neck was not resected completely. Histopathological examination revealed retinoblastoma of the orbit and the parotid and submandibular glands. After chemotherapy and additional local radiotherapy on the parotid and submandibular glands, the tumour was inactive and stable. CONCLUSIONS: Delayed detection and inappropriate management contribute to poor outcomes. Fundus examinations, education regarding the early signs of RB, and optimization of the therapeutic strategy for RB may play important roles in ocular health.

Recurrent Retinoblastoma With Mandibular Metastasis: A Case Report.

Retinoblastoma (RB) is an aggressive intraocular tumor arising from cells of the retina. Infrequently, distant metastasis occurs in advanced stages of the disease. This case report describes the metastasis of the tumor to the mandible, an extremely rare phenomenon. This condition was observed for a recurrent tumor in a patient who initially underwent chemotherapy, making this case report unique and providing new insights into the behavior of this tumor. An overview of the management of a metastatic RB also is discussed.

Does Medullary Versus Cortical Invasion of the Mandible Affect Prognosis in Patients With Oral Squamous Cell Carcinoma?

PURPOSE: Whether mandibular involvement by oral squamous cell carcinoma (OSCC) could be identified as a factor for cancer staging and prognosis prediction remains a subject of debate. In addition, the influence of different types of mandibular invasion (cortical or medullary invasion) on patients' prognosis remains unclear. The aim of this systematic review was to establish whether mandibular invasion or its subset should be considered an independent prognostic factor for patients with OSCC. MATERIALS AND METHODS: The search for eligible studies was performed according to the predesigned inclusion criteria for a systematic review. Mandibular invasion and invasion depth were considered the primary and secondary predictor variables, respectively. The electronic search was performed using 12 databases. Manual searching covered 14 related journals and references of the included studies were scanned. The risk of bias assessment was evaluated by 2 reviewers using risk-of-bias assessment tools recommended by Saltaji et al (Angle Orthod 82:1115, 2012). Two reviewers extracted the data in duplicate. RevMan 5.2 was used for meta-analysis to assess the primary outcomes (disease-free survival and overall survival) and the secondary outcomes (2- and 5-year survival rate and local control). RESULTS: Eighteen studies (total, 3,756 participants) were included and used as the study sample. Among these included studies, 7 had an unclear risk of bias and the remaining showed a high risk. The results of the meta-analyses showed a significant relation between mandibular invasion and overall survival (P = .04) and, most importantly, that medullary involvement (P = .0001), but not cortical involvement (P = .66), could decrease overall survival. When focusing on disease-specific survival, mandibular medullary involvement predicted a poor disease-specific survival (P < .0001), but cortical involvement showed no effect (P = .66). CONCLUSION: This review showed that OSCC mandibular medullary invasion, and not mandibular invasion or mandibular cortical invasion, could be an independent prognostic factor for patients.

Melanoma Metastasis to the Mandible-Case Report and Comprehensive Literature Review.

With the incidence of melanoma increasing yearly, there is a need for heightened awareness of its metastatic potential and for screening with appropriate referral for identification of pre-metastatic lesions. Melanoma has a 16- to 25-month period to metastasize from a localized disease to one with a median survival no longer than 12 months once metastasis occurs. Nearly one third of oral metastases are found to be the first indication of occult malignancy from a distant site. This report describes the case of a 54-year-old woman with a longstanding undiagnosed acral lentiginous melanoma with metastasis to the maxillofacial region. She underwent resection and died 7 months later. The authors analyzed case reports and the current literature for biological mechanisms of metastasis, risk factors, clinical presentation, classifications, staging, treatment modalities, prognosis, and current therapy modalities.

Metastasis of Mammary Gland Malignant Phyllodes Tumor to the Mandibular Region: A Case Report and Review of the Literature.

Phyllodes tumor is a rare breast tumor described by Müller (1938) as a lesion comprising leaflike stromal fibrous components and narrow cysts. The frequency of distant metastasis from this entity is reportedly approximately 20%, and no effective therapy has been established, so the prognosis is poor. This report describes the case of a 60-year-old woman with a history of left lung resection who showed metastasis of a mammary gland malignant phyllodes tumor to the oral cavity. Intraoral examination showed an elastic, hard mass measuring 28 × 27 mm in the gingiva around the left mandibular second molar. Biopsy examination showed growth of giant cells and roughly circular cells showing positivity for S-100, p63, and vimentin on immunohistochemical staining. The authors diagnosed metastasis of the mammary gland malignant phyllodes tumor to the left mandible and performed cyber knife irradiation (44 Gy in 5 fractions) of the left mandible. The mass in the oral cavity disappeared after cyber knife irradiation, but the patient died of direct invasion to the spine.

Metastatic follicular thyroid carcinoma: a case report.

OBJECTIVE: A case of oral metastatic follicular thyroid carcinoma is presented. BACKGROUND: Metastatic tumours are more frequent in older individuals and can be the only sign/symptom of an undiagnosed primary malignancy. CASE REPORT: A 69-year-old lady presented with an enlarging mandibular swelling. Incisional biopsy and imaging studies were suggestive of metastatic follicular thyroid carcinoma. CONCLUSION: Metastatic tumours should be considered in the differential diagnosis of mandibular swellings especially in older individuals.

Unusual Presentation of an Adenocarcinoma of the Lung Metastasizing to the Mandible, Including Molecular Analysis and a Review of the Literature.

Lung cancer is the most frequent cause of cancer-related death worldwide. Metastases of non-small cell lung carcinoma to the oral and maxillofacial region are rare. Thus, the diagnosis of a metastatic lesion in the oral cavity is challenging to the clinician and to the pathologist. This report presents a case of a 72-year-old man with metastatic lung adenocarcinoma located in the posterior mandibular region. Next-generation sequencing analysis showed no important mutations in the relevant genes except in the TP53 tumor suppressor gene.

Recurrent Malignancy in Osteoradionecrosis Specimen.

PURPOSE: Osteoradionecrosis (ORN) is a well-known complication of head and neck radiation therapy. Statistically, the mandible is the most commonly affected site. The incidental finding of malignancy in the resection specimen has been documented but is somewhat rare. The aim of this review is to investigate the presence of recurrent carcinoma and sarcoma or new primary malignancies in resection specimens previously diagnosed and treated as ORN. PATIENTS AND METHODS: This study is a retrospective case series. We conducted a chart review of all cases managed at the University of Miami Miller School of Medicine/Jackson Memorial Hospital. The inclusion criteria included a history of head and neck carcinoma treated with radiation of at least 6,000 cGy; clinical diagnosis of ORN; and surgical intervention with osseous resection for treatment of ORN. The study endpoint measured included microscopic evidence of malignancy in the resected ORN specimen. Additional data collected included gender, age, and type of primary pathology. RESULTS: A total of 564 patients met the inclusion criteria. Of these patients, 14 had microscopic evidence of cancer in the specimen (2.48%) and 5 had a proven second primary malignancy in the foregut (1 in the lung, 0.18%, and 4 in the oropharynx, 0.70%). In 1 of the 14 patients, a high-grade sarcoma was diagnosed and the patient died within 1 year of diagnosis. In the treatment of our ORN patient population, a total of 19 malignancies were found collectively (3.37%). Of the 564 patients, 352 were men and 212 were women. The median age was 46 years (range, 33 to 97 years). Head and neck squamous cell carcinoma represented 531 cases in our sample, followed by 28 cases of salivary gland carcinoma and only 5 cases of sarcoma. CONCLUSIONS: Although the finding of malignancy in ORN patients is relatively rare (3.37% in this study), oral and maxillofacial surgeons should be cognizant of its potential presence. The treatment of malignant disease is different than that of ORN, and a multidisciplinary treatment approach is recommended if a malignancy is diagnosed in an ORN patient.

Metastatic rectal adenocarcinoma in the mandibular gingiva: a case report.

BACKGROUND: Oral metastatic tumor from a rectal adenocarcinoma is very uncommon. The primary site is usually assumed based on the past clinical history. In the case of oral metastatic tumors, they commonly have a poor prognosis because often they have already spread to other sites. CASE PRESENTATION: We present the case of a 64-year-old male patient with secondary metastasis to the mandibular gingiva via lung metastasis after the surgical resection of a primary rectal adenocarcinoma. The gingival lesion grossly appeared as a swollen mass, making mastication difficult. The patient received palliative radiotherapy for the mandibular mass lesion. However, tumor reduction was accompanied by the development of pneumonia and deterioration of the patient's cachexia. Thus, the radiotherapy was discontinued but the patient died 2 months postradiotherapy. In the long term after its primary resection, the rectal adenocarcinoma was deduced to have finally metastasized to the oral region. CONCLUSIONS: In this case, we consider a distant secondary metastasis to the oral region from a rectal malignancy. In such cases, careful clinical and pathologic evaluations are necessary, with careful consideration of the inclusion of palliative treatment in the therapeutic management.

Pre-surgical assessment of mandibular bone invasion from oral cancer: comparison between different imaging techniques and relevance of radiologist expertise.

PURPOSE: To compare diagnostic performance between computed tomography (CT) and magnetic resonance imaging (MRI) for the detection of bone infiltration from oral cancer, and to test interobserver agreement between radiologists with different expertises. MATERIALS AND METHODS: Pre-surgical CT and MRI were reviewed independently by two radiologists with different expertises in head and neck oncology. A third radiologist reviewed CT and MRI simultaneously. Interobserver agreement was calculated by Cohen test. Association between radiological evidence of bone infiltration and histological reference was tested by Fisher's exact test or Chi-squared test, as appropriate. Receiving operator curve was calculated and area under the curve (AUC) was compared between CT, MRI, and both methods together. RESULTS: Interobserver agreement was moderate: the trainee under-reported periosteal reaction on CT and inferior alveolar canal involvement on MRI. Imaging findings associated with histologic evidence of bone infiltration were: periosteal reaction and cortical erosion on CT; bone marrow involvement, contrast enhancement within bone; and inferior alveolar canal involvement on MRI. Sensitivity of MRI alone (74 %) was higher than CT (52 %). Simultaneous review of CT and MRI showed the highest specificity (91 %), with the increase of diagnostic performance in the subgroup of subjects with positive MRI (AUC = 0.689; p = 0.044). CONCLUSION: Higher expertise allows pre-surgical detection of clinically relevant signs of bone infiltration sensitivity of MRI alone is higher than CT for the detection of bone infiltration from oral cancer. In MRI positive cases, diagnostic integration with combined review of CT and MRI is suggested for optimal diagnostic performance.

A case of retinoblastoma metastasizing to the mandible and review of literature.

OBJECTIVES: The aim of this case report and review was to determine the characteristics of retinoblastoma. METHODS: One case report was introduced along with previous reports on retinoblastoma metastasizing to the mandible. RESULTS: Sixteen cases from 14 reports were included in this study. Including the present case, 11 of 16 patients died within 8 months. DISCUSSION: Retinoblastoma rarely metastasizes to the mandible. However, metastasis to other organs should be considered, and specialists should be consulted if retinoblastoma metastasis to the mandible is observed. Moreover, it is necessary to follow up patients after multidisciplinary therapy is completed, because subsequent complications of the teeth and jawbones associated with therapy could occur.

[Jawbone metastasis masquerading as dental pain].

Metastases to the oral cavity are rare. However, in 25% of cases, oral symptoms will be the first sign of metastatic disease. The incidence of jaws metastases is twice as high as the incidence of metastases to the soft tissues of the oral cavity. In some cases, jaws metastases can mimic dental or periodontal pain. We report a case of a 67 year old female who was referred to our clinic because of severe pain on her left posterior mandible which was not relieved by endodontic treatment of the first and second molar. She was diagnosed with breast cancer in 2005 and had been treated with surgery, chemotherapy and radiotherapy. Seven years later, lung metastases were found and she was treated with chemotherapy. Later on, brain metastases developed which had been treated with radiotherapy. On presentation, she complained of pain on the posterior left mandible which was accompanied by a burning sensation of the lower left lip and chin. CT scan revealed a soft tissue mass perforating the lingual and buccal plates of the posterior left mandible, which was compatible with a diagnosis of metastasis. Radiotherapy rapidly relieved the pain. Unfortunately, the patient passed away one month later. Dentists should be able to recognize the signs and symptoms associated with metastases to the jaws and should include it in the differential diagnosis, especially in patients with oncologic background.

Emission Computed Tomography for the Diagnosis of Mandibular Invasion by Head and Neck Cancers: A Systematic Review and Meta-Analysis.

PURPOSE: To detect the diagnostic efficacy of emission computed tomography (ECT) in detecting mandibular invasion caused by head and neck cancers. MATERIALS AND METHODS: Thirteen databases were searched electronically to retrieve studies for inclusion and a manual search also was conducted. Study inclusion, data extraction, and quality assessment were completed by 2 reviewers independently. Meta-DiSc 1.4 and STATA 11.0 were used to conduct the meta-analysis. RESULTS: Seventeen studies involving 668 participants were included. One study had a low risk of bias, 2 had a high risk, and the rest had unclear risk. Meta-analysis showed that for the diagnosis of mandibular invasion single-photon ECT (SPECT) had a mean sensitivity (SEN) of 0.96, a mean specificity (SPE) of 0.66, an area under the curve (AUC) of 0.8989, and a Q* (point on the summary reviewer operator characteristic curve when SEN equaled SPE) of 0.8300. Positron emission tomography combined with computed tomography (PET/CT) had a mean SEN of 0.83, a mean SPE of 0.90, an AUC of 0.9290, and a Q* of 0.8640. The comparison between the diagnostic efficacy of SPECT and PET/CT showed that SPECT was superior for SEN (P = .0014) and PET/CT had a significantly better SPE (P = .001). The summary diagnostic efficacy between these modalities did not differ significantly (P > .05). CONCLUSIONS: The present clinical evidence showed that SPECT is an excellent tool to exclude patients with no mandibular invasion, but is not as good as PET/CT to confirm the diagnosis.

Open Dislocation of the High Ankle Joint After Fibular Graft Harvesting.

The free microvascular fibula and soft tissue transfer has become a widely used method for reconstruction of different regions. Donor site morbidity for free fibula microvascular flaps has generally been reported to be low, or at least acceptable. We describe the case of a patient who underwent vascularized free fibula graft harvest for mandibular reconstruction. After 21 months, he had sustained an open dislocation of the left high ankle joint during recreational sports activity. We did not found such case in the published data.