Large Palatal Pleomorphic Adenoma Complicated by Dysphagia: A Case Report.

A 60-year-old Japanese woman presented with a palatal pleomorphic adenoma measuring 5 cm in size. In addition to impairments during the oral preparatory and oral transport phases, dysphagia with nasopharyngeal closure disorder was observed in the pharyngeal phase. After resection of the tumor, dysphagia resolved, and the patient was immediately able to eat a standard meal. A videofluoroscopic swallowing study confirmed improvement in the movement of the soft palate compared with the pre-operative condition.

Selective deep lobe parotidectomy via retroauricular hairline (Roh's) incision for deep lobe parotid pleomorphic adenoma.

OBJECTIVE: Deep lobe parotid tumour is commonly removed with the covering superficial lobe of parotid gland. Total or subtotal parotidectomy leads to an increase in surgical morbidity. This study evaluated recurrence and function after selective deep lobe parotidectomy via retroauricular hairline (Roh's) incision for pleomorphic adenoma. MATERIALS AND METHODS: Twenty-eight patients with deep lobe parotid pleomorphic adenomas underwent selective deep lobe parotidectomy with preservation of the superficial lobe and the facial lobe via Roh's incision. Each patient was evaluated with any complications, cosmetic and salivary functions and local recurrence. RESULTS: Superficial lobe-preserving surgery via Roh's incision was successfully applied to all patients without injury to the facial nerve and the Stensen's duct for a median operation time of 65 min. Facial nerve paralysis was found only temporarily in 9 (32%) patients, and other complications were minimal. None of the patients had postoperative Frey's syndrome. Salivary secretory function in the operated side was well preserved. No recurrence was found in the patients for a median follow-up of 94 months. CONCLUSIONS: Selective deep lobe parotidectomy via Roh's incision is a reliable option of treatment for deep lobe parotid pleomorphic adenoma.

[Unexpected cause of tracheal obstruction: pleomorphic adenoma with lipometaplasia]. / Adenoma pleomorfo con lipometaplasia: inesperada causa de obstrucción traqueal.

Pleomorphic adenoma of the trachea is a benign tumor, extremely rare, that generates obstructive symptoms and sometimes is confused with asthma, which delays its diagnosis. We present the case of a 40-year-old woman with history of asthma and airway obstruction, of 8-months duration. Fibrobronchoscopy showed polypoid, pedunculated lesion in first tracheal ring which occluded 80% of the lumen, that was resected. The histopathological diagnosis was pleomorphic adenoma with lipometaplasia. Treatment of these tumors consists in complete resection of the lesion and long-term follow-up due to low probability of recurrence, malignancy and metastasis.

El adenoma pleomorfo de tráquea es un tumor benigno, infrecuente, que genera cuadros obstructivos, pudiendo confundirse con asma, lo que retrasa su diagnóstico. Presentamos el caso de una mujer de 40 años con antecedentes de asma y episodios de obstrucción respiratoria de 8 meses de evolución. La fibrobroncoscopía mostró una lesión polipoide, pediculada, en el primer anillo traqueal, con oclusión de 80% de la luz, que se resecó. El diagnóstico histopatológico fue adenoma pleomorfo con lipometaplasia. El tratamiento de estos tumores consiste en la resección completa de la lesión y seguimiento a largo plazo por la baja probabilidad de recurrencia, malignización y metástasis.

A Rare Co-Existence of Multiple Cerebral Cavernous Malformations and Pleomorphic Adenoma of the Parotid Gland in Myotonic Dystrophy.

Myotonic dystrophy is an autosomal dominant inherited disorder primarily affecting muscle function. Myotonia, progressive muscle weakness and wasting, and associated systemic involvement, i.e., cataracts, cardiac conduction defects and endocrine abnormalities especially insulin resistance, are the characteristic features. Recent evidence has shown an increased risk of developing benign as well as malignant tumours in such patients. We report a 39-year male of myotonic dystrophy who presented with multiple cerebral cavernous malformations in addition to pleomorphic adenoma of the parotid gland. Though the association of myotonic dystrophy with salivary gland neoplasms has been sparsely documented in the literature, but the co-existence with multiple cerebral cavernous malformations has not been reported so far. Our case is the first of its kind. Key Words: Cerebral cavernous malformations, Myotonic dystrophy, Parotid gland, Pleomorphic adenoma.

Chronic Granulomatous Dacryoadenitis Associated With Pleomorphic Adenoma.

Chronic granulomatous inflammation occurs rarely alongside pleomorphic adenomas of the major salivary glands but would not appear to have been reported with lacrimal gland adenomas. We describe the clinical features, imaging and histopathology for 4 patients (3 female) who had granulomatous inflammation alongside lacrimal gland adenomas-the patients being with age 39, 44, 48, and 53 years at time of surgery. One patient had an asymptomatic lesion found on imaging, and the other 3 had symptoms for between 3 years and several decades. Conjecturally, this rare phenomenon might arise from an inflammatory response to leakage of secretions from the ductular components of the glands.

[The influence of the capsular defect as a risk factor for recurrence in pleomorphic adenoma]. / Der Einfluss des Kapseldefektes als Risikofaktor für die Rezidivbildung beim pleomorphen Adenom der Glandula parotidea.

OBJECTIVE: Aim of this study was to enlighten various aspects of the capsular defect of a pleomorphic adenoma and to analyze its influence on recurrence rate with the help of our data and the relevant literature. MATERIAL AND METHODS: The clinical records and histopathological findings of all patients who underwent parotidectomy for pleomorphic adenoma between 2005 and 2020 were evaluated retrospectively. The histological slides of all tumours were critically re-evaluated for this study by an experienced head and neck pathologist. The records of the cases with a capsular defect were examined for information on age, gender, type of surgery and surgical outcome. RESULTS: A total of 845 patients were included in the primary group. 7 (0.8 %) recurrences were detected, all of these were resected without a capsular rupture. Positive margins were found in 37 cases (4.4 %). In 13/37 cases the capsular defect was detected intraoperatively, whereas the defect was only identified histopathologically in 24 of the 37 cases. In the group of the intraoperative capsular opening, with a median follow-up of 62 months, the sonographic examination suspected a unilocular recurrence of the pleomorphic adenoma in two cases. CONCLUSION: The intraoperative capsule opening does not inevitably seem to lead to a pre-programmed recurrence.

Primary De novo ductal adenocarcinoma of the lacrimal gland.

BACKGROUND: Primary ductal adenocarcinoma of the lacrimal gland is a rare and aggressive malignant epithelial lacrimal gland neoplasm, morphologically and phenotypically resembles salivary duct carcinoma, and both strongly resemble infiltrating ductal carcinoma of breast. METHOD: Retrospective Chart review of cases of malignant lacrimal gland tumors from 2013 July to 2020 July. Authors describe the clinico radiological, morphological and immunohistochemical features of primary ductal adenocarcinoma (PDA) of lacrimal gland. Extensive review of literature of PDA of lacrimal gland and salivary gland ductal carcinoma has been performed. RESULTS: Retrospective chart review of the last 7 years yielded 22 malignant lacrimal gland neoplasms of which 4 cases demonstrated features of primary ductal adenocarcinoma of lacrimal gland, 2/4 cases showed an evidence of a pre existing pleomorphic adenoma and 2 were found to be de novo ductal adenocarcinomas. PDA of lacrimal gland showed expression of CK7, CK19, AR, HER2, cyclin D1 and were negative for CK5/14, CK 20, ER, PR, PSA, TTF-1, S-100 and SMA. Expression of GCDFP-15 was noted in one case. The presence of multiple events of loco-regional recurrences and/or distant metastasis necessitated a multidisciplinary approach. CONCLUSIONS: Authors have expressed the need of clinical correlation; thorough tissue sampling and extensive immunohistochemical work up in identification of de novo PDA's and their molecular subtypes. A multi-institutional study might help in formulating the diagnostic criteria, identification of actionable targets, and thus study the role of targeted therapy in this rare and aggressive tumor which may result in better patient outcomes.

The significance of tyrosine kinase receptor B and brain-derived neurotrophic factor expression in salivary duct carcinoma.

Salivary duct carcinoma (SDC) is a high-grade salivary gland neoplasm. It may occur de novo or secondarily from pleomorphic adenoma (ex-PA), with secondary development accounting for more than 50% of the cases. In recent years, the expression of tyrosine kinase receptor B (TrkB), which is in the same family as HER2, has been confirmed in various types of carcinomas. However, there are a few studies on SDC. In order to examine the expression and role of TrkB in SDC, we investigated it. Immunohistochemistry was used to detect the expression of TrkB and its ligands, brain-derived neurotrophic factor (BDNF) and neurotrophin-4 (NT-4) in 20 patients with SDC. The mRNA levels of TrkB, BDNF, and NT-4 were analyzed using quantitative polymerase chain reaction. TrkB was negative in 10 cases and positive in 10 cases, BDNF was negative in 11 cases and positive in 9 cases, and NT-4 was positive in all cases. There was a high number of TrkB-positive cases in the pT4 group and The H-score of TrkB was also significantly higher in the stage III and IV groups. There was a high number of BDNF-positive cases in the ex-PA group and Histo-score of BDNF had a trend of high expression in ex-PA. There were no significant differences or correlations in mRNA expression. Our results suggest that TrkB may be involved in SDC tumor growth.

Myotonic dystrophy and recurrent pleomorphic adenomas: Case report and association hypothesis.

We report a case of a patient with concurrent myotonic dystrophy and recurrent pleomorphic adenoma and hypothesize the association between both diseases. A 58-year-old man with classic myotonic dystrophy type 1 was diagnosed with pleomorphic adenoma. Appropriate treatment was commenced. Massive recurrences occurred within 15, 28 and 22 months respectively, after repeated surgical removal. Three case reports on similar occurrences of synchronous myotonic dystrophy and pleomorphic adenoma are discussed and an association between both disease entities is hypothesized. A conceivable association between myotonic dystrophy and pleomorphic adenoma is hypothesized by upregulation of the Wnt/Beta-catenin signaling pathway, initiated by a decreased expression of microRNA, pleomorphic adenoma gene 1 induced Beta-catenin accumulations and alterations in tumor suppressor genes and oncogenes due to RNA processing defects induced by the expanded repeat in the DMPK gene.

Adenoma pleomorfo originado en pared lateral nasal: reporte de un caso clínico / Pleomorphic adenoma of lateral nasal wall: a case report

RESUMEN El adenoma pleomorfo constituye la neoplasia benigna más frecuente de las glándulas salivales mayores, y puede también presentarse en otros sitios con mucha menor frecuencia como orofaringe, hipofaringe y nasofaringe. El adenoma pleomorfo intranasal es muy infrecuente y los casos descritos en la literatura local se refieren a tumores septales. Se presenta un caso clínico de una paciente que consulta por obstrucción nasal unilateral a derecha asociado a, epistaxis y epífora ipsilateral con estudio imagenológico y biopsia que sugiere adenoma pleomorfo de la pared lateral nasal. Se realiza revisión bibliográfica al respecto.

ABSTRACT The pleomorphic adenoma is the most frequent benign neoplasm of the major salivary glands. It can also present itself in other places with much less frequency such as oropharynx, hypopharynx and nasopharynx. The intranasal pleomorphic adenoma is very unusual and the cases described in the local literature address septal tumors. A clinical case is presented of a patient who consulted for unilateral right nasal obstruction associated with epistaxis and ipsilateral epiphora with imaging study and biopsy suggesting pleomorphic adenoma of the lateral nasal wall. A bibliographic review is made in this regard.

Giant De Novo Pleomorphic Adenoma of Parapharyngeal Space, Can It Cause Spinal Deformity? A Case Report.

STUDY DESIGN: Case report. INTRODUCTION: De novo giant pleomorphic adenoma is a rare tumor of the parapharyngeal space (PPS). Tumors of the PPS can grow to a large size, compromising the space of the upper aerodigestive tract. However, involvement of the paravertebral region is unexpected. In extremely exceptional circumstances, these tumors can produce spinal deformity. CASE REPORT: A 25-year-old man presented with a longstanding mass in the neck and oral cavity. He had complaints of dysphagia, snoring, and restricted neck movements because of the large size of the tumor. Imaging showed a large mass occupying the PPS extending to the paravertebral region and causing deformity of the cervical spine. Excision was done uneventfully via a minimal access transcervical approach. Residual spinal deformity dealt with postoperative physiotherapy with improvement in lordosis and lateral tilt. No tumor recurrence occurred till 26 months of follow-up. CONCLUSION: Tumors of the PPS can grow to a large size and involve the neck and PPS, even causing spinal deformity. Careful evaluation is required for ascertaining the origin of the tumor and deciding the treatment plan.

Sinonasal pleomorphic adenoma: A single institution case series combined with a comprehensive review of literatures.

OBJECTIVE: This study aimed to reveal diagnosis, management, and treatment outcome characteristics of sinonasal pleomorphic adenoma and to identify predictors for disease recurrence. METHODS: This study comprised a retrospective review of data from seven patients with sinonasal pleomorphic adenoma who were treated by the authors at a tertiary medical center between January 2005 and May 2017. In addition, the MEDLINE database was searched for literature published in English. A systematic review of the published articles was performed. RESULTS: A total of 42 articles were eligible for review. Data from the seven treated patients and 101 patients described in the literature were included in the analysis. An endoscopic approach was employed in all cases treated at our institution, and recurrence was found in one patient with a large, 5.5-cm tumor. In reviewing prior publications, the mean age at surgery was 45.4 years, and the primary sign at diagnosis was nasal obstruction. The most common primary tumor site was the nasal septum (57.4%), and the mean tumor size was 3.6cm (range, 0.5-10cm) in 76 cases for which detailed information was available. Endoscopic endonasal resection achieved successful oncologic control in applied cases (25/25, 100%). Recurrence was found in eight of 101 patients. Multivariate analysis identified tumor origin of the paranasal sinus (adjusted OR 31.000, p=0.016) as being significantly associated with a high prevalence of recurrence. CONCLUSION: Pleomorphic adenoma is a rare benign tumor of the sinonasal tract. Surgery that achieves clear margins is the treatment of choice. The tumor may be accessible via an endoscopic endonasal approach in most cases, although incomplete resection of the tumor stem may allow for tumor recurrence. In addition, our findings suggest that a tumor originating from the paranasal sinus is significantly associated with a high prevalence of recurrence.

Metastasizing pleomorphic adenoma in the multiple organs: A case report on FDG-PET/CT imaging.

RATIONALE: Pleomorphic adenoma, the most common tumor of the salivary glands, is usually benign. It is well known, however, that pleomorphic adenomas occasionally undergo malignant transformation to carcinoma ex pleomorphic adenoma and can metastasize. More rarely pleomorphic adenomas can metastasize without histological malignant transformation. We herein report an unusual case of pleomorphic adenoma with multiple metastases comprehensively demonstrated on F-18 fluorodeoxyglucose positron emission tomography/computed tomography (FDG-PET/CT). PATIENT CONCERNS: A 63-year-old woman was referred to our hospital because of urine occult blood and lung nodules detected on a health checkup. She had a history of resection of a pleomorphic adenoma in the parotid gland at the age of 40 years and tumor recurrence at the age of 53 years. CT scan and magnetic resonance imaging revealed bilateral renal tumors, multiple pulmonary nodules, and an osteolytic lesion in the first lumbar vertebra. DIAGNOSES: Ultrasonography-guided percutaneous biopsy of the right renal tumor revealed a myxoid epithelial tumor that was consistent with metastasis of the pleomorphic adenoma from the parotid gland. INTERVENTIONS: The patient was carefully observed with regular imaging examinations. OUTCOMES: The multiple lesions gradually progressed, and FDG-PET/CT subsequently revealed additional metastases in the liver and perineum. LESSONS: Metastases of pleomorphic adenomas may occur years after the initial disease in association with local recurrences. Careful observation with whole-body imaging such as FDG-PET/CT is necessary.

Syncope caused by a pleomorphic adenoma: Case report and literature review.

Pleomorphic adenomas are considered the most common salivary gland tumors, although they rarely occur in the parapharyngeal space. To the best of our knowledge, this is the first case report of a parapharyngeal parotid pleomorphic adenoma causing syncope. A 57-year-old man was admitted for left-sided blurred vision, left-sided weakness, dysarthria, lightheadedness, and syncope. Upon his admission, an electrocardiogram showed sinus bradycardia, and computed tomography of the neck with contrast showed a large parapharyngeal mass involving the prestyloid compartment, leading to compression of blood flow through the internal carotid artery. The mass was biopsied via intraoral fine-needle aspiration, which revealed cytology consistent with pleomorphic adenoma. The mass was resected via a transcervical approach, and a total parotidectomy was performed. The patient's hypotensive and bradycardic episodes disappeared after surgery. Surgical pathology showed a benign pleomorphic adenoma with a hemorrhagic and necrotic center and without capsular invasion or malignant transformation.

Metastasising pleomorphic adenoma of the submandibular salivary gland presenting with spinal cord compression: case report and review of cases with CNS involvement.

We present a very rare case of a metastasising pleomorphic adenoma with spinal cord compression and review the small handful of cases with central nervous system (CNS) involvement in the literature. This case is unusual as most other reported cases are preceded by local recurrences; also, there is only one previous case of an MPA presenting with spinal cord compression. Metastasising pleomorphic adenoma is a rare malignant salivary gland tumour which, despite "benign" histological appearances produces secondary deposits in distant sites.