Metastatic merkel cell carcinoma to the thyroid gland: Case report and review of the literature.

BACKGROUND: Merkel cell carcinoma (MCC) is an aggressive and rare neuroendocrine tumor, accounting for less than 1% of skin cancers. Metastasis primarily manifests in the cervical lymph nodes but rarely affect the thyroid. METHODS: We report a case of primary head and neck cutaneous MCC with metastasis to the thyroid gland. A review of the literature of MCC with thyroid metastasis was conducted. RESULTS: We identified five cases of MCC with thyroid metastasis. Primary sites included the distal upper and lower extremities, axilla, buttock, and groin. Treatment courses varied including thyroidectomy, immunotherapy, and expectant palliative measures. Time from initial diagnosis to thyroid metastasis ranged from four months to four years. Tissue diagnosis was achieved in 5 of 6 cases. CONCLUSIONS: MCC with thyroid metastasis is rare and likely represents aggressive disease. Despite advances in treatment and surveillance, outcomes for MCC remain poor. Ongoing research may establish predictors for treatment response.

Challenges in the Management of Symptomatic Fallopian Canal Meningoceles: A Multicenter Case Series and Literature Review.

OBJECTIVE: To describe the presentations, the diagnosis, our treatment approaches, and the outcomes for 11 patients with fallopian canal meningocele (FCM). STUDY DESIGN MULTICENTER: Retrospective case series. SETTING: Tertiary referral centers. PATIENTS: Patients (N = 11) with radiographically or intraoperatively identified, symptomatic FCM. INTERVENTIONS: Surgical repair of cerebrospinal fluid (CSF) leak and meningocele versus observation. MAIN OUTCOME MEASURES: Presentation (including symptoms, radiographic imaging, and comorbidities), management (including surgical approach, technique for packing, use of lumbar drain), clinical outcomes (control of CSF leak, meningitis, facial nerve function), and revision surgery. RESULTS: Patients presented with spontaneous CSF leak (n = 7), conductive (N = 11) and sensorineural hearing loss (n = 3), nonpositional intermittent vertigo (n = 3), headaches (n = 4), and recurrent meningitis (n = 1). Risk factors in our series included obesity (n = 4), Chiari 1 malformation (n = 1), and head trauma (n = 2). Noncontrast computed tomography of the temporal bone and magnetic resonance imaging were positive for FCM in 10 patients. Eight patients were managed surgically via a transmastoid approach (n = 4), combined transmastoid and middle fossa (N = 3), or middle fossa alone (n = 1); three were managed conservatively with observation. Postoperative complications included worsened facial nerve palsy (n = 1), recurrent meningitis (n = 1), and persistent CSF leak that necessitated revision (n = 1). CONCLUSIONS: Facial nerve meningoceles are rare with variable presentation, often including CSF otorrhea. Management can be challenging and guided by symptomatology and comorbidities. Risk factors for FCM include obesity and head trauma, and Chiari 1 malformation may present with nonspecific otologic symptoms, in some cases, meningitis and facial palsy. Layered surgical repair leads to high rates of success; however, this may be complicated by worsening facial palsy.

Diagnostic Difficulties of Plunging Ranula: A Review of 18 Cases.

OBJECTIVE(S): Despite advancements in imaging techniques and cytological analysis, plunging ranula remains a challenging surgical, radiologic, and pathologic phenomenon. Of the 18 patients we evaluated at our institution, we highlight three cases that illustrate the high rate of misleading imaging and cytological results when assessing plunging ranula. METHODS: Imaging results, biopsy findings, operative techniques, and pathological reports were reviewed from patients who had either a preoperative or postoperative diagnosis of ranula and underwent surgery by a single head and neck surgeon at a tertiary care center. RESULTS: Of the 18 identified patients, computed tomography was correct on preoperative imaging 73% of the time and magnetic resonance imaging was correct on preoperative imaging 71% of the time. Two patients underwent preoperative ultrasound and their ultrasound reports did not accurately diagnose the presence of a ranula. Two patients underwent preoperative fine needle aspiration biopsy due to inconclusive preoperative imaging, in which results suggested either a ranula or epidermal cyst. Both ultimately did not match the final pathology. Three of eighteen patients (17%) underwent an inappropriate initial surgery due to incorrect imaging diagnoses and/or biopsy findings. CONCLUSION: Despite use of preoperative modalities to distinguish plunging ranula from other cystic floor of mouth lesions, surgeons must be aware that no workup modality is fully precise. The potential for revision surgery must be included in all preoperative discussions for presumed plunging ranula. LEVEL OF EVIDENCE: 4 Laryngoscope, 134:2689-2696, 2024.

Thyroid cancer necrosis not evident on imaging: A cautionary case series on poorly differentiated thyroid carcinoma diagnosed only on final pathology.

OBJECTIVE: Poorly-differentiated thyroid cancer (PDTC) is a highly aggressive malignancy which is recently defined and understudied in the radiologic literature. Necrosis is a key histopathologic criterion for the diagnosis of PDTC. We illustrate the current difficulty in accurate identification of histopathologic necrosis on preoperative imaging. METHODS: A series of seven patients with the final diagnosis of PDTC from our institution were identified. Multimodality preoperative imaging was analyzed by two head and neck radiologists. Final pathology reports were queried confirming histopathologic evidence of necrosis. RESULTS: Patients presented with a wide range of preoperative imaging features. A consistent imaging appearance confirming necrosis was not identified. All patients were subsequently upstaged to PDTC following final pathological analysis. CONCLUSION: A lack of definitive evidence of necrosis on preoperative imaging does not exclude the possibility of PDTC. We demonstrate the need for further research to establish a clear methodology for the preoperative diagnosis of PDTC.

Report of a vagal paraganglioma at the cervicothoracic junction.

BACKGROUND: Vagus nerve paragangliomas are rare tumors, comprising 0.03% of head and neck neoplasms. These tumors are usually located cephalad to the hyoid bone, and there is only one previously reported case that arose from the lower third of the neck. METHODS: We describe the second reported case of a lower neck vagus nerve paraganglioma that was managed with a limited sternotomy for access and surgical removal. RESULTS: A 66-year-old male presented with a long-standing lesion of the cervicothoracic junction. CT, MRI, and Ga-68 DOTATATE PET/CT showed an avidly enhancing 5.2 × 4.2 × 11.5 cm mass extending from C6 to approximately T4 level. FNA confirmed the diagnosis. The patient underwent catheter angiography and embolization via direct puncture technique followed by excision of the mass via a combined transcervical and limited sternotomy approach. CONCLUSION: We describe an unusual case of vagal paraganglioma at the cervicothoracic junction with retrosternal extension requiring a sternotomy for surgical excision.

Snapshot of proteomic changes in Aspergillus oryzae during various stages of fermentative processing of pea protein isolate.

Pea (Pisum sativum) is one of the most abundant and sustainable alternate source of protein. Although pea proteins have good quantities of most of the essential amino acids, they have a limited supply of tryptophan, methionine and cysteine. Moreover, pea proteins have poor techno-functional properties compared to proteins from animal sources, limiting their use in certain food applications. Bioprocessing techniques like solid-state fermentation (SSF) and enzymatic processing have been explored to improve the nutrient profile and functionality of pea proteins. However, there is a lack of information about proteomic changes in the food matrix during fermentation of the pea substrate. In this research, samples during SSF of pea protein isolate with Aspergillus oryzae were used for shotgun mass spectrometry (LC-MS/MS) analysis to identify the underlying functional pathways which play direct or indirect roles in enabling the colonization of the substrate leading to potential improvement of functional and nutritional value of pea protein. Results revealed the identity of A. oryzae proteins involved in different metabolic pathways that differed during various stages of SSF. Among them, methionine synthase was identified as an abundant protein, which catalyzes methionine biosynthesis. This might suggest how fermentation processes could be used to improve the presence of sulfur containing amino acids to rebalance the essential amino acid profile and improve the nutritional quality of pea proteins.

A Case of HPV+ Squamous Cell Carcinoma of the Perigeniculate Area and Middle Fossa.

Herein we present an unusual case of a primary HPV+ perigeniculate, extra-axial middle fossa skull base tumor and our management thereof. Laryngoscope, 133:3158-3160, 2023.

Extratumoral invasion: A unique phenomenon of aggressive recurrent oropharyngeal squamous cell carcinoma.

BACKGROUND: Oropharyngeal squamous carcinomas cause significant morbidity and mortality. Poor prognosticators include lymphovascular and perineural invasion. Extratumoral phenotypes of these histologic findings confer worse prognoses. METHODS: We report eight cases of recurrent oropharyngeal cancer with diffuse extratumoral lymphovascular invasion (ELVI) or extratumoral perineural invasion (EPNI) and review the existing literature. RESULTS: On salvage resection for recurrence following primary radiation or chemoradiation, six patients manifested ELVI and two showed EPNI. These patterns conferred difficulty with complete surgical clearance; final pathologic analysis demonstrated positive margins for all eight patients. The six patients with ELVI were p16+ and the two with EPNI were p16-. Currently, two patients are deceased and six patients are alive at an average follow-up of 17.4 months. Of the six living patients, 2 have a new recurrence and are in hospice while 4 have no evidence of disease. CONCLUSIONS: ELVI and EPNI have received little consideration in the literature as unique histopathologic features of oropharyngeal squamous carcinoma. We present the first series on these adverse extratumoral features in recurrent disease. We call attention to these unique histologic features in the setting of recurrent oropharyngeal cancer to encourage others to track the results of therapeutic intervention and to identify successful strategies for treatment.

A rare case of canalicular adenoma in the parotid gland: Highlighting diagnostic limitations of fine-needle aspiration.

BACKGROUND: Canalicular adenoma is a rare, benign tumor of primarily salivary gland origin that presents mostly in the upper lip. However, there are only six reports in the English literature detailing canalicular adenoma of the parotid gland, none of which discuss discrepancy between preoperative cytology and surgical pathology. In this report, we present a rare case of parotid gland canalicular adenoma where preoperative ultrasound-guided fine-needle aspiration (USFNA) suggested malignancy. The patient was treated with deep lobe parotidectomy due to the FNA results and her multiple comorbidities. However, her tumor may have been treated with observation alone if canalicular adenoma had been suspected prior to surgery. MAIN FINDINGS: A 59-year-old female with a history of heart and lung disease presented with a 1.6 cm well defined, enhancing lesion involving the superficial portion of the right parotid gland. This lesion was incidentally noted on CT angiography (CTA) of the neck and chest. The well-defined characteristics of this lesion on CT imaging suggested benign neoplasm. However, USFNA results were suggestive of a malignant parotid lesion. The patient subsequently underwent right deep lobe parotidectomy with facial nerve dissection and superficial musculoaponeurotic system (SMAS) rotational flap reconstruction. Surgical pathology and immunohistochemistry yielded a final diagnosis of benign canalicular adenoma. CONCLUSIONS: USFNA diagnosis of CA is extremely difficult due to its low-grade neoplastic cells mimicking neoplastic cells in other benign and malignant tumors of the head and neck. FNA remains a useful tool for assessing malignancy risk, but the results always have some level of uncertainty and do not provide sufficient detail. Therefore, FNA results should be interpreted in concert with imaging and patients' medical history. Cytopathologists can also report salivary gland FNA results in a more uniform and detailed manner by utilizing the Milan System for Reporting Salivary Gland Cytopathology (MSRSGC).

A novel technique for management of stenosis of the postlaryngectomy stoma with preservation of a functional tracheoesophageal puncture following tracheal resection.

Tracheostomal stenosis following total laryngectomy presents a serious concern for surgeons and patients. Although various techniques correct tracheostomal stenosis, none address an existing tracheoesophageal puncture (TEP). We present an approach to repair tracheostomal stenosis, requiring tracheal resection, and preserve the TEP in a functional position needed for speech rehabilitation. A 62-year-old male with squamous carcinoma of the right true vocal fold underwent a salvage total laryngectomy with placement of a tracheoesophageal prosthesis. Seven years later, he developed tracheal narrowing. A tracheal resection and tracheoplasty were performed to manage the stenosis while maintaining the tracheoesophageal puncture. Six months postoperatively, the patient was well, with no stomal narrowing or trend toward collapse and uneventful healing. He achieved fluent voice easily with stomal occlusion. We present a novel surgical technique to correct for tracheostomal stenosis following total laryngectomy. Our technique allows for TEP preservation to facilitate speech rehabilitation postoperatively.

Oncocytoid Variant of High-Grade Salivary Duct Carcinoma: A Potential Diagnostic Pitfall. A Report on Two Patients.

Salivary duct carcinoma (SDC) is an uncommon and aggressive salivary malignancy. The oncocytoid variant of salivary duct carcinoma (OSDC) has only been reported in the English literature once before. Here we detail two new patients. A 71-year-old female presented with a painless enlarging left parotid mass. Imaging and fine-needle aspiration were nondiagnostic. The second patient, a 79-year-old male, presented with painless swelling in the right cheek. Imaging was nondiagnostic. Both patients underwent surgical resection. Histopathology revealed bland yet infiltrative OSDC in both cases. These tumors were AR+ (androgen receptor) by immunohistochemistry. Potential difficulty exists in distinguishing the oncocytoid variant of SDC, a rare and relatively bland tumor, from oncocytoma, a more commonly encountered entity. AR expression can aid in the correct diagnosis.

Cuneiform (cuniculatum) variant of oral squamous cell carcinoma revisited: Presentation of two cases and literature review of a rare entity.

BACKGROUND: Oral carcinoma cuniculatum (OCC) is a rare, locally aggressive tumor, which tends to invade underlying bone. We present two cases of OCC, one demonstrating invasion of the mandible and the other limited to the tongue. METHODS: An 87-year-old male presented with a right-sided buccogingival lesion. Biopsy results led to a diagnosis of verrucous hyperplasia, which was later revised to OCC. Additionally, a 94-year-old female presented with a left lateral tongue lesion. A biopsy showed in-situ and invasive keratinizing squamous cell carcinoma that was later defined as a soft tissue OCC. RESULTS: Following surgical resection, the diagnosis of OCC was established in both patients. We provide a comprehensive literature review of OCC in the context of both case presentations. CONCLUSIONS: OCC is a rare entity, which has a tendency to be misdiagnosed. We emphasize the importance of recognizing the common features of OCC in order to aid in accurate diagnosis.

Adenoid Cystic Carcinoma of the Gingiva: A Case Report and Literature Review.

OBJECTIVES: Adenoid cystic carcinoma (ACC) is a commonly encountered salivary gland malignancy. However, it rarely occurs in the gingiva, an area generally thought to be devoid of minor salivary glands. We present a case occurring in this unusual site and review other reported cases. METHODS: A 56 year-old male presented with a right-sided mandibular toothache for 1 year and underwent dental extraction. Due to persistent pain, follow up examination revealed a large gingival lesion. A biopsy was positive for adenoid cystic carcinoma. RESULTS: The patient underwent a complete right segmental mandibulectomy and was reconstructed with a fibular osteocutaneous free flap. Three months postoperatively, during the planning for adjuvant radiation therapy, the patient developed pain in the left mandible. Imaging revealed extensive involvement of the left native mandible. Deep bone biopsies in several areas of the left mandible revealed ACC. He then underwent a complete left hemi-mandibulectomy and reconstruction with a fibular osteocutaneous free flap. Tensor fascia lata suspension slings were placed due to concern for an open mouth deformity attributable to disruption of bilateral masticator slings. He will undergo adjuvant radiation therapy. Our review of the literature revealed 50 cases of gingival ACC published since 1972. Disease recurrence and distant metastases were noted in several patients, occurring at the latest after 30 years follow-up. CONCLUSIONS: Given its indolent behavior, high proclivity for late recurrence and metastasis, and overall infrequency, ACC represents a pathology that requires early diagnosis and comprehensive long-term surveillance. While ACC is well described in oral cavity sites with high densities of minor salivary glands, it is not commonly seen in the gingiva. As such, gingival ACC may display a unique biological and/or clinical character. We offer the first literature review of this rare entity.

Rare case of anaplastic plasmacytoma in the sinonasal tract.

BACKGROUND: Extramedullary plasmacytomas are tumors that develop from plasma cells and rarely express anaplastic features. To our knowledge, there have only been three reported cases of anaplastic plasmacytomas of the sinonasal tract in the English literature. We detail the fourth case. METHODS: A 70-year-old male was seen with a 4-month history of nasal congestion, bloody mucous, and left sided nasal obstruction. On positron emission tomography/computed tomography, the lesion was FDG-avid with an SUVmax of 25.1. A biopsy of the lesion and subsequent immunohistochemical staining confirmed the diagnosis of an anaplastic plasmacytoma. RESULTS: The patient is undergoing a 5-week course of curative-intent radiation therapy. CONCLUSION: Extramedullary plasmacytomas with anaplastic features are very rare. We highlight the value of thorough histopathological review and detailed immunostains to arrive at a diagnosis of anaplastic extramedullary plasmacytoma.

Reconstruction of the external auditory canal: The tragal flap revisited and review of contemporary reconstructive techniques.

BACKGROUND: Primary malignancies arising in the external auditory canal (EAC) are rare and usually are treated surgically. We review techniques to reconstruct the EAC following ablative surgery, and introduce a rarely utilized tragal skin flap which has particular advantages for reconstruction of limited anterior EAC defects. METHODS: The terms "tragal flap", "external auditory canal", "preauricular tragal flap", "reconstructive techniques" were searched on PubMed and Google Scholar. RESULTS: Our review identified one description of a tragal flap to reconstruct the EAC following resection of a malignancy. We add an additional case of a preauricular tragal flap to reconstruct the anterior EAC following resection of a recurrent basal cell carcinoma located in the EAC that led to a circumferential defect. CONCLUSION: There are several surgical techniques that can be utilized to reconstruct the EAC. We describe a novel tragal flap used to reconstruct the anterior EAC following resection of a recurrent tumor.

Diagnosis and management of pharyngoesophageal stenosis: A comprehensive approach to prophylactic, endoscopic, and reconstructive treatment options.

BACKGROUND: Pharyngoesophageal stenosis (PES) is a serious complication that substantially impacts functional outcomes and quality of life (QOL) for up to a third of head and neck cancer patients who undergo radiotherapy. Dysphagia is often multifactorial in nature and is a devastating complication of treatment that impacts patients' QOL, general health and overall wellbeing. The authors detail the clinical presentation, risk factors, imaging characteristics, preventive measures, and multimodality treatment options for PES. METHODS: The authors present a comprehensive management algorithm for PES, including treatment by dilation, stenting, spray cryotherapy and dilation, and reconstructive treatment options utilizing different pedicled and free flaps. RESULTS: The authors advocate for a thorough assessment of the extent and degree of pharyngoesophageal involvement of PES to determine the optimal management strategy. CONCLUSIONS: The development of post treatment dysphagia requires appropriate imaging and biopsy, when indicated, to rule out the presence of persistent/recurrent cancer. Multidisciplinary management by a team of physicians well-versed in the range of diagnostic and therapeutic interventions available for PES is critical to its successful management.

Unusual location of a second branchial cleft cyst presenting in the suprasternal notch.

BACKGROUND: Second branchial cleft cysts (SBCCs) are congenital benign tumors that comprise up to 90% of all branchial cleft anomalies. SBCCs typically present in the lateral neck along the anterior border of the upper third of the sternocleidomastoid muscle. We describe a case of a SBCC presenting in an unusual location in the lower neck close to midline. METHODS: An 18-year-old male presented with a 2-year history of a neck mass in the suprasternal notch. Imaging findings were reviewed with a head and neck radiologist who felt that the findings were highly suggestive of a fourth branchial cleft cyst. RESULTS: The patient underwent surgical excision of the mass. Final pathologic evaluation confirmed the diagnosis of a second branchial cleft cyst. CONCLUSIONS: Though extremely uncommon, second branchial cleft cysts can extend to the suprasternal notch and should not be excluded from the differential diagnoses of lower neck masses.

Multifocal Parotid Oncocytic Cystadenomatosis: A Rare Process that Can Mimic Malignancy.

Multifocal cystic oncocytosis (MCO) is a rare, benign process accounting for approximately 0.1% of salivary gland lesions. Salivary oncocytosis is characterized by multiple unencapsulated solid nodules of oncocytic cells derived from transformed striated ducts. MCO is a variant of salivary oncocytosis which manifests as cystically dilated striated ducts. It is difficult to obtain a definitive preoperative diagnosis of MCO; therefore, these lesions are commonly treated with surgery. We report the unique case of a 66-year-old male who previously underwent a superficial left parotidectomy for a pleomorphic adenoma. Four years later, he presented with clinical and radiographic suspicion of a multifocal recurrent pleomorphic adenoma. The patient subsequently underwent a revision parotidectomy. However, final pathology confirmed a diagnosis of MCO. Although MCO is commonly treated with surgery due to lack of a definitive preoperative diagnosis, surgery is unnecessary outside of diagnostic, functional or cosmetic considerations. Thus, if a patient with parotid oncocytosis treated by superficial parotidectomy develops disease re-manifestation in the residual deep lobe, further surgery is not indicated. There is no risk of malignant progression in this process. We report on this unusual entity as it may mimic salivary malignancy or, as in this case, recurrence of benign disease.

Metastatic thyroid carcinoma to the parapharyngeal and retropharyngeal spaces: Systematic review with seven newly reported cases describing an uncommon presentation of a common disease.

BACKGROUND: Metastatic thyroid carcinoma to retropharyngeal and parapharyngeal (RP/PP) lymph nodes is rare. Literature suggests previous lateral neck dissection (LND) may alter patterns of lymphatic drainage in the neck, predisposing to these less common sites of spread. METHODS: PRISMA-guided systematic search for all published cases detailing RP/PP metastases of well-differentiated thyroid carcinoma from 1970 to 2019. RESULTS: Seventy articles were identified and 44 were included, along with seven cases treated at our institution, totaling 239 cases. Cases represented both retropharyngeal (60.7%) and parapharyngeal (39.3%) metastases identified in the initial (27.6%) and recurrent (72.4%) setting. CONCLUSION: RP/PP metastases generally present in the recurrent setting. RP/PP metastases often represent high-risk disease, and surgical treatment is recommended.