Osteoradionecrosis of the hyoid bone complicated by pharyngocutaneous fistula: A case report and literature review.

BACKGROUND: Osteoradionecrosis (ORN) is a well-known complication following irradiation of head and neck malignancies. ORN commonly occurs in the mandible but is rarely reported in the hyoid bone. CASE PRESENTATION: A 76-year-old female with a history of oropharyngeal squamous cell carcinoma presented with pharyngocutaneous fistula 14 years after primary chemoradiation. Imaging showed necrosis of the hyoid bone. She underwent excision of the hyoid to rule out malignancy. Pathology was negative for carcinoma, but did show extensive fragmentation and bony necrosis consistent with ORN. The patient's clinical course, surgical treatment, and management considerations are discussed here. CONCLUSIONS: Hyoid ORN should remain in the differential during diagnostic workup of previously irradiated head and neck cancer patients. The presentation of a pharyngocutaneous fistula should prompt workup to rule out malignancy before assigning a diagnosis of ORN.

Primary mucinous eccrine carcinoma of the buccal space: A case report and review of the literature.

IMPORTANCE: Mucinous eccrine carcinoma is a rare entity that most commonly affects the head and neck. Due to its low frequency of occurrence, review of its etiology, histopathology, and treatment strategies is beneficial to all clinicians who may encounter similar appearing masses. OBSERVATION: An 84-year-old male presented with a blue mass on the left cheek. This mass started as a small bump and grew significantly over one year. His primary care physician monitored its growth and ultimately referred to an otolaryngologist. Imaging findings revealed a multi-lobular solid and cystic left buccal lesion. FNA was suggestive of low grade mucoepidermoid carcinoma. INTERVENTION: Patient underwent surgical excision with primary closure of the defect. Frozen section was consistent with low grade salivary malignancy. Final pathology revealed primary mucinous eccrine carcinoma of the skin. CONCLUSIONS AND RELEVANCE: Mucinous eccrine carcinoma is a rare entity commonly seen in the head and neck region. Mucinous deposits to the skin from primaries elsewhere in the body are much more common than primary lesions of the skin. Histology is a key component of the diagnosis but full oncologic workup is required. Treatment typically includes wide local excision with possible adjuvant chemotherapy or radiation for high risk features.

Septic arthritis of the sternoclavicular joint: A unique late complication after tracheostomy.

BACKGROUND: Septic arthritis of the sternoclavicular joint is a rare infection associated with significant morbidity and mortality. Several risk factors for septic arthritis have been reported in the literature ranging from immunodeficiency to intravenous drug use. CASE PRESENTATION: A 63-year-old male previously treated for synchronous squamous cell carcinomas of the epiglottis and floor of mouth presented with tenderness and swelling of the sternoclavicular joint two months after tracheostomy decannulation. Computed tomography and bone scans confirmed the diagnosis of septic arthritis of the sternoclavicular joint. The patient's clinical course, surgical treatment, and management considerations are discussed here. CONCLUSION: Septic arthritis of the SCJ is a rare but serious infection. Once diagnosed, septic arthritis of the SCJ should be promptly treated to prevent further morbidity and mortality.

Ipsilateral Ophthalmic and Cerebral Infarctions After Cosmetic Polylactic Acid Injection Into the Forehead.

A 55-year-old woman developed no light perception vision in her right eye 5 days after an injection of polylactic acid cosmetic filler into her right forehead. Diffuse corneal edema and anterior chamber inflammation prohibited any view to the posterior segment to identify the cause of her profound vision loss. MRI of the orbits with diffusion-weighted imaging showed hyperintensity of the right optic nerve with signal reduction on apparent diffusion coefficient mapping, consistent with ischemia. Our patient also was found to have acute infarctions in the distribution of the right anterior cerebral artery on MRI of the brain despite having no permanent focal neurologic deficits aside from vision loss.

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.

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.

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.

Oral squamous carcinoma: Aggressive tumor pattern of invasion predicts direct mandible invasion.

BACKGROUND: Aggressive histologic worst pattern of invasion (WPOI) in surrounding soft tissue has been shown to be predictive of higher local recurrence and poorer survival in oral cavity squamous cell carcinoma (OCSCC) patients. This study investigates whether aggressive WPOI can predict the mandibular invasion phenotype. METHODS: Patients consecutively diagnosed with OCSCC undergoing a mandibulectomy (marginal or segmental) between 2013 and 2018 were reviewed. Senior physicians re-reviewed radiologic scans and pathologic slides of 44 cases. RESULTS: Aggressive WPOI (WPOI-4, 5) is significantly associated with infiltrative bone invasion. Non-aggressive WPOI (WPOI-1, 2, 3) is significantly associated with the absence of bone invasion. CONCLUSIONS: WPOI has become a useful tool that further characterizes the biologic behavior of OCSCC. Potentially, planned surgery may escalate from a marginal to segmental mandibulectomy based on aggressive WPOI for patients with radiographically uncertain cortical status. Further studies are needed to validate the relationship between OCSCC WPOI and mandible status.

Intraoral sebaceous carcinoma metastatic to the lung and subcutis: Case report and discussion of the literature.

BACKGROUND: Intraoral sebaceous carcinoma is a rare form of sebaceous carcinoma with only 9 published cases in the world literature to date. We present a 10th case of intraoral sebaceous carcinoma located in the anterior maxillary gingiva with metastases to the lung and subcutis and discuss 3 possible etiologies for this unique presentation. METHODS: We analyze the clinical presentation, pathology, histology, and genetic testing for a single case study and review relevant literature. RESULTS: The histologic findings of the lung tumor and surgical excisions of the tumors in the gingiva and subcutis suggest the gingiva is the primary site. There is no evidence for the genetic abnormalities consistent with Muir-Torre syndrome. CONCLUSION: The histologic findings suggest the oral cavity is the most likely site of tumor origin. This is the first case of intraoral sebaceous carcinoma reported to arise in the gingiva as well as to spread to cutaneous sites.

Pediatric odontogenic fibromyxoma of the mandible: Case report and review of the literature.

BACKGROUND: Odontogenic fibromyxoma is an uncommon benign tumor arising from the maxillofacial bones. Fibromyxomas are rare in children under 10 years of age. Although this tumor is reported most frequently in the mandible for the general population, it has rarely been reported in the mandible in children <10 years of age. METHODS: We reviewed the 39 reported cases of odontogenic fibromyxoma in children under the age of 10. We add 1 case to the literature. RESULTS: This case represents the seventh case of odontogenic fibromyxoma of the mandible in a child under the age of 10 years reported in the English literature. CONCLUSION: A case of pediatric odontogenic fibromyxoma in the mandible is described. Although rare in the pediatric population, odontogenic fibromyxomas should be included as a differential diagnostic consideration when evaluating tumors of the maxillofacial skeleton. Accurate pathologic diagnosis is critical to ensure proper management.

Asymptomatic lower extremity deep venous thrombosis resulting in fibula free flap failure.

OBJECTIVES/HYPOTHESIS: The successful harvest and transplant of a fibular flap depends on many factors, including healthy inflow and outflow systems. A contraindication to harvesting a fibular flap is disease of the lower extremity arterial system; therefore, preoperative evaluation of the arterial system is routine. Preoperative evaluation of the venous system is not routine, unless there is clinical suspicion of venous disease. METHODS: Retrospective chart review. RESULTS: Two cases of occult deep venous thrombosis (DVT) were encountered intraoperatively resulting in nontransplantable flaps. CONCLUSIONS: This finding represents a serious concern, and we believe that venous imaging should be considered in patients with significant risk factors for harboring an occult DVT. Laryngoscope, 2009.