Base of tongue metastasis of cutaneous malignant melanoma with rhabdoid and neuroendocrine features: Report of a rare case and review of the literature.

Metastatic malignant melanoma (MM) represents a highly aggressive cancer associated with overall poor prognosis. Various anatomic sites can be affected, including the oral cavity and the oropharynx. It may mimic other entities by assuming a variety of clinical appearances and exhibiting a plethora of microscopic variations. Herein, we present a case of a 63-year-old male with a MM metastasizing to the base of tongue, which developed 5 years after the original diagnosis and treatment of cutaneous MM of the chest and heralded its relapse; subsequently, neurological symptoms developed as a result of metastasis to the brain. Diagnostic challenges were encountered, as the tongue lesion clinically masqueraded as a pedunculated reactive lesion and microscopically displayed unusual rhabdoid and neuroendocrine features. Tumor cells expressed S-100, HMB-45, Melan-A, and SOX-10, while most cells with rhabdoid morphology were also positive for myogenin and Myo-D1. Chromogranin and synaptophysin positivity was further noticed in a subset of cells, suggestive of focal neuroendocrine differentiation. Molecular investigation revealed mutations for the BRAF V600E gene. Divergent differentiation of tumor cells may cause diagnostic pitfalls necessitating thorough immunohistochemical analysis. The presence of rhabdoid features and neuroendocrine differentiation are very uncommon, while their co-existence is extremely rare. Better characterization of such microscopic variations in MMs with evaluation of their potential biologic significance is warranted.

Oncological outcomes in patients undergoing major glossectomy for advanced carcinoma of the oral tongue.

INTRODUCTION: Major glossectomy is the treatment of choice in locally advanced tongue cancer. It remains the only option in the presence of recurrent or residual disease. The long-term outcomes for patients undergoing major glossectomy have traditionally been poor, with significant morbidity and poor oncological outcomes. The aim of this study was to report on oncological outcomes in patients undergoing major glossectomy. METHODS: All patients undergoing major glossectomy between 2014 and 2018 were included in the study. The data of 85 patients with advanced carcinoma of the oral tongue were evaluated. All were under the care of a single surgical and reconstructive team at two hospitals in Mumbai. RESULTS: The median patient age was 45 years. At the most recent follow-up, 55 patients (65%) were alive, 47 of whom were disease free. Twenty-nine patients (34%) had locoregional recurrence and twenty-five (29%) had distant metastasis. At a median follow-up of 19 months, rates for 2-year locoregional control, disease free survival (DFS) and overall survival (OS) were 69%, 61% and 62% respectively. Perinodal extension demonstrated a trend towards poor DFS (p=0.060), as did perineural invasion (p=0.055). Node positivity was a significant factor for poor OS, DFS and locoregional control. Multiple node involvement was significantly associated with poor OS on multivariate analysis (p=0.002). CONCLUSIONS: Node positivity and multiple node involvement were associated with poor outcomes. Major glossectomy may be offered as a curative option for selected patients with advanced carcinoma of the oral tongue with node negative or limited neck nodal disease (N1).

Transoral robotic surgery for the benefit of patients with head and neck cancer of unknown primary: our experience at St George's University Hospital, London.

INTRODUCTION: Head and neck carcinoma of unknown primary represents 1-5% of all head and neck cancers and presents a diagnostic and therapeutic dilemma. In approximately 40% of cases, a primary tumour location remains unknown despite investigation. With advancements in our understanding of the role of high-risk human papilloma virus in head and neck cancer, transoral robotic surgery presents an option for diagnosis and therapy. MATERIALS AND METHODS: This is a retrospective case series from a single centre. Case notes were reviewed for 28 patients who had transoral robotic surgery for head and neck carcinoma of unknown primary between May 2015 and July 2019. RESULTS: Transoral robotic surgery identified an oropharyngeal primary tumour in 19 of 28 (67.8%) patients. All oropharyngeal primaries were p16 positive. The base of tongue identification rate was 63.2%. Median length of inpatient stay postoperatively was 1.0 day. Normal oral intake resumed within 48 hours in 96% (27/28) of patients. Three patients (10.3%) suffered minor postoperative bleeds that were all managed conservatively. DISCUSSION: The base of tongue primary identification rate (63.2%) in this series is consistent with that previously reported (43-63%; 95% confidence interval). Primary tumour identification rate if a patient is p16 positive is 86.3% (19/22), with 100% of these being oropharyngeal. We suggest future investigation into p16 status as a means of stratifying patients with head and neck carcinoma of unknown primary for transoral robotic surgery. CONCLUSION: Transoral robotic base of tongue mucosectomy (or lingual tonsillectomy) is a promising technique that offers a high yield of positive identification for the primary tumour. It is well tolerated with minimal associated morbidity. Our findings are comparable with those in the current literature.

HPV insertional pattern as a personalized tumor marker for the optimized tumor diagnosis and follow-up of patients with HPV-associated carcinomas: a case report.

BACKGROUND: In clinical oncology, only a few applications have been developed using HPV as a personalized tumor marker, a lack most probably related to the limited information obtained by the classical Polymerase Chain Reaction (PCR) approach. To overcome this limitation, we have recently developed the capture-based Next-Generation Sequencing (NGS) "CaptHPV" assay, designed to provide an extensive and comprehensive molecular characterization of HPV DNA sequences associated with neoplasias, ie the sequence of the viral genome (245 genotypes), its physical state, viral load, integration site and genomic alterations at integration locus. These data correspond to highly specific tumor markers that can be used to improve diagnosis and patient's follow-up. CASE PRESENTATION: We report here a case that is a straightforward and practical illustration of the power of the CaptHPV method. A patient developed successively a carcinoma of the anal canal and of the tongue. The two tumors were squamous cell carcinoma, found associated with HPV16 using PCR. In order to document a possible metastasis to the tongue from the anal cancer, we performed CaptHPV analysis on the two tumors. The analysis of the anal carcinoma found 55 viral/human hybrid reads allowing the identification of the HPV16 DNA integration in the 4q25 chromosomal band locus with a 178,808 bp deletion in the cell genome. Molecular analysis of the tongue tumor disclosed 6110 reads of HPV16, with a viral pattern strictly identical to that of the anal tumor. A total of 131 hybrid reads between HPV16 and the cell genome were found, corresponding exactly to the same locus of integration of viral DNA at the 4q25 site. The 178,808 bp genomic deletion was also found in the lingual tumor. The exact identity of HPV insertional signatures in the two tumors, demonstrates unambiguously that the tongue tumor derived from the anal cancer whereas neither histological immunophenotyping nor classical viral analysis using PCR could allow a definitive diagnosis. CONCLUSION: Our observation indicates that the establishment of a detailed cartography of HPV DNA sequences in a tumor specimen provides crucial information for the design of specific biomarkers that can be used for diagnostic, prognostic or predictive purposes.

Elective Neck Dissection for Management of Early- Stage Oral Tongue Cancer

Background: The occult neck metastasis rate is very high with tongue cancers. The aim of this study was to assess the current role of elective neck dissection (END) in management of early-stage oral tongue cancer with a focus on lymph node metastasis. In addition, effects of END on regional or systemic disease recurrence and survival were investigated. Methods: This retrospective study included patients with stage I and II tongue cancer recruited from our National Cancer Institute (NCI) over a time period of six years (2007-2013). The collected data were analyzed for disease free survival (DFS) and recurrence rate. Results: A total of 144 patients presented to our NCI with oral tongue cancer but only 88 were staged clinically and radiologically as early stage (stage I, stage II). Some 53% were smokers. Most lesions were dealt with by surgery, either by wide local excision (22%) or hemiglossectomy (78%). Treatment of neck lesions was either by neck dissection (85.2%) or "wait and see" (14.8%). The rates for local and nodal recurrence were 7.9% and 20.4%, respectively. Analysis of associations between DFS and different factors revealed significance for adoption of adjuvant therapy and the dissected lymph node status. Conclusion: Controversy still exists regarding neck management.

Tongue metastasis as an initial presentation of renal cell carcinoma.

Aim Distal metastases to oral cavity are very rare. Adenocarcinoma, renal cell carcinoma (RCC) and squamous cell carcinoma are the most common metastatic tumours. The aim of this study was to assess the diagnostic relevance of metastatic renal cell carcinoma on tongue manifestations in large patient series. Methods A patient with distal RCC metastases to the tongue and neck lymph nodes as the first manifestations of malignancy was presented. Also, all reports described during 106-year period relating to distal RCC metastasis to the tongue were evaluated. Results In the presented patient, initial presentation of RCC was metastasis to the tongue. Three months after nephrectomy, tongue tumour resection and radical neck dissection were performed. Histopathological analysis confirmed RCC metastasis. Postoperatively, the patient underwent radiotherapy and polychemotherapy. Tongue metastasis from RCC is extremely rare with only 51 cases reported during the 106-year period. In only 7 patients tongue metastasis was reported as an initial presentation of RCC. Conclusion The RCC metastasizing to the tongue is extremely rare and thus it poses a diagnostic challenge especially when it is the first sign of malignancy. Therapeutic decisions should maximize patient comfort and minimize morbidity considering the poor long-term prognosis.

Lingual metastasis as an initial presentation of renal cell carcinoma: a case report.

BACKGROUND: Renal cell carcinoma is the third most common tumor that metastasizes to the head and neck, after breast and lung carcinomas. Tongue metastasis as an initial presentation of renal cell carcinoma is extremely rare, and very few cases have been reported. The prognosis is poor. We present a rare case of metastatic renal cell carcinoma that initially presented as a tongue lesion. CASE PRESENTATION: We report the case of a 55-year-old white man who presented with a large exophytic lesion on his tongue. A biopsy was taken, and pathologic examination showed a poorly differentiated carcinoma including a sarcomatoid component. Subtotal glossectomy with neck dissection were planned, but a positron emission tomographic-computed tomography scan showed a left kidney mass. Immunohistochemical evaluation of the tongue lesion was performed, and it was compatible with metastasis from primary renal cell carcinoma. The biopsy of the renal lesion showed a high-grade unclassified renal cell carcinoma. Although our patient underwent systemic therapy, he died of hemorrhagic complications 3 months after the initiation of therapy. CONCLUSION: Tongue lesions require a complete assessment to distinguish a metastasis from a primary cancer in order to give the appropriate treatment.

A case report of tongue metastasis from lung squamous cell carcinoma and literature review.

RATIONALE: Tongue metastasis from lung cancer is extremely rare, and the prognosis of these patients is rather poor. PATIENT CONCERS: A 56-year-old man was found a 4-cm cavity lesion in the left upper lobe, which was initially misdiagnosed as tuberculosis. DIAGNOSES: A case of lung squamous cell carcinoma that metastasized to the base of a patient's tongue. INTERVATIONS: We send the biopsy of the lung and the tongue lesions for gene sequencing. OUTCOMES: He received systemic chemotherapy, but continued to have pain at the base of his tongue and died 7 months later. LESSONS: From sequencing data, mutations in KRAS proto-oncogene, GTPase (KRAS), phosphatidylinositol-4,5-bisphosphate 3-kinase catalytic subunit alpha (PIK3CA), and tumor protein p53 (TP53) were found in the tumor biopsy of the patient. All of these were indicators of poor prognosis.

Metastatic chordoma of the tongue: Case report.

Chordomas are rare bone tumors that arise from notochord remnants. They most commonly occur in the sacrum, but they also can be seen in the skull base, cervical spine, and thoracolumbar vertebrae. Chordomas account for 1 to 4% of all primary skeletal tumors. They are usually indolent, locally growing tumors. Distant metastasis has been reported in 3 to 48% of cases. When metastasis occurs, it is usually observed in the lung, bone, and liver. To the best of our knowledge, no case of a chordoma metastasis to the tongue has been previously reported in the literature. We report such a case in a 61-year-old man.

Successful facial artery pseudoaneurysm coiling and pedicle preservation following free tissue transfer.

Patients undergoing free tissue reconstruction are at risk for development of an anastomotic pseudoaneurysm, which may present as delayed neck hemorrhage or a pulsatile neck mass. Diagnosis may be achieved by noninvasive imaging, angiography, and exploration. Management strategies for head and neck pseudoaneurysms have included open vessel ligation, open direct vessel repair, endovascular parent vessel embolization, and, most recently, endovascular pseudoaneurysm embolization. In patients with anastomotic pseudoaneurysms where adequate flap inosculation is doubted, endovascular pseudoaneurysm embolization with pedicle preservation may be an appropriate primary treatment approach. We discuss the successful endovascular coiling of an external carotid artery branch anastomotic pseudoaneurysm in a patient one month after free tissue reconstruction of a total laryngopharyngectomy and partial glossectomy defect.

Oral and gastrointestinal symptomatic metastases as initial presentation of lung cancer.

Metastasis to the tongue, duodenum or pancreas from primary lung cancer is uncommon. Primary lung cancer presenting with symptoms related to metastases at these sites, at initial presentation is extremely rare. We report a 45-year-old man with disseminated lung malignancy who presented with dyspepsia, melena, symptoms due to anaemia and swelling in the tongue. Oral examination revealed a hard submucosal anterior tongue lesion. Biopsies from the tongue lesion and the duodenal ulcer seen on upper gastrointestinal endoscopy were suggestive of metastasis from lung primary. CT revealed lung primary with disseminated metastasis to lung, liver, adrenals, kidneys, head and body of pancreas, duodenum and intra-abdominal lymph nodes. The patient was treated with palliative chemotherapy. The unusual presentation and diagnostic details are discussed.

[Metastasis to the Tongue from Renal Cell Carcinoma 10 Years after Nephrectomy : A Case Report].

A 71-year-old woman underwent left radical nephrectomy for renal cell carcinoma (clear cell carcinoma, pT1bN0M0) ten years previously. She noticed a tumor on the tip of her tongue and was admitted for dental and oral surgery. The tumor was about 10 mm in size, and tumor resection was done. It was pathologically diagnosed as clear cell carcinoma, which was metastasis of renal cell carcinoma. Computer tomography scan during the same period revealed left hilar lymph node and bilateral lung metastases. We chose to use sunitinib as the treatment for the metastases. Computer tomography revealed a complete response (CR) after sunitinib treatment was given for 10 months, and we are still continuing the treatment to maintain the CR status.

High-grade urothelial carcinoma with squamous differentiation metastasizing to the tongue.

Tumors metastasizing to the head and neck region are uncommon. Metastasis of urothelial carcinoma to the maxillofacial region is exceedingly rare and mostly involves the jaw. We present a case of urothelial carcinoma metastasizing to the tongue. Immunohistochemistry in conjunction with fluorescent in situ hybridization was used to confirm the relation between the primary and metastatic lesions, making it the first such reported case employing the UroVysion (Catalogue number 02 J27-025, Abbott Molecular Inc., Des Plaines, IL, USA) fluorescent in situ hybridization probe in a metastatic lesion in the head and neck region.

[The Combination Therapy of Gemcitabine Plus Paclitaxel Induced Complete Response in a Tongue Skin Brain Metastasis of Bladder Carcinoma: A Case Report].

Tongue, skin and brain metastases of bladder cancer are very rare and few cases have been reported. We report a case of tongue, skin and brain metastases of bladder cancer. A 61-year-old woman was referred to our hospital with gross hematuria. Transurethral resection of the bladder (TURBT), tongue biopsy and skin biopsy were performed. Pathological findings showed urothelial carcinoma, G2, micro papillary variant, pT2＞ and tongue and skin metastases from urothelial carcinoma of bladder. After three cycles of chemotherapy (gemcitabine plus paclitaxel), tongue and skin metastases disappeared. Cystoscopy revealed no tumor of bladder. Eleven months later, she was admitted to our hospital because of disturbance of consciousness. Magnetic resonance imaging (MRI) showed multiple brain metastases. Rechallenge of chemotherapy (gemcitabine plus paclitaxel) restored from disturbance of consciousness and MRI showed partial response of brain metastases. We performed six additional courses of chemotherapy. Skin, tongue and brain metastases from bladder cancer indicate poor risk. Chemotherapy (gemcitabine plus paclitaxel)could be effective against these matastases.

Computerized tomography based tumor-thickness measurement is useful to predict postoperative pathological tumor thickness in oral tongue squamous cell carcinoma.

BACKGROUND: Tumor thickness has been shown in oral tongue squamous cell carcinoma (OTSCC) to be a predictor of cervical metastasis. The postoperative histological measurement is certainly the most accurate, but it would be of clinical interest to gain this information prior to treatment planning. This retrospective study aimed to compare the tumor thickness measurement between preoperative, CT scan, and surgical specimens . METHODS: We retrospectively included 116 OTSCC patients between 2001 and 2013. Thickness was measured on computer tomography imaging and again surgical specimens. RESULTS: The median age was 66 years. 62.8 % of patients were smokers with a mean of 31.4 pack-years. Positive nodal disease was reported in 41.2 %. Mean follow-up time was 33.1 months. The correlation between CT scan-based tumor thickness and surgical specimens based thickness was significant (Spearman rho = 0.755, P < 0.001). CONCLUSION: Tumor thickness assessed by CT scan may provide an accurate estimation of true thickness and can be used in treatment planning.

Tongue metastasis of melanoma.

Malignant melanoma may metastasize to virtually any organs. Metastases to the oral cavity are uncommon and tongue metastases are very rare. The patient was a 79-year-old woman with cutaneous malignant melanoma. Lesion was widely excised and lymph node dissection was performed. She also had adjuvant chemotherapy. She is the unique malignant melanoma case who had recurrence on tongue 10 years later in literature. In conclusion, the prognosis of patients with tongue metastasis is poor and most of these patients have underlying widespread diseases.

Soft Tissue Sarcoma With Tongue Metastasis: A Case Report and Literature Review.

Soft tissue sarcomas are rare malignant tumors that develop from mesenchymal cells. Metastasis is predominantly hematologic, with the lungs being the most common site. Metastasis to the oral cavity is a rare occurrence. The most common primary tumors to metastasize to the oral cavity are adenocarcinoma of the lung, breast, and kidney. This report describes a case of a 41-year-old man who was diagnosed with myxofibrosarcoma of the lower extremity and underwent neoadjuvant chemoradiation followed by surgical resection. Two years later, he presented with metastasis to the tongue and lungs. The literature on tongue metastasis of soft tissue sarcoma is reviewed and discussed. Surgeons providing care to patients with a soft tissue sarcoma should maintain a strong clinical suspicion for distant metastases in patients with this type of tumor.