A case of renal sarcoidosis complicated by parotid gland and uterine lesions.

BACKGROUND: Sarcoidosis is a systemic disease that can affect multiple organs. While pulmonary sarcoidosis is most commonly observed, renal sarcoidosis occurs less frequently. We herein report a case of sarcoidosis with an exceptionally rare distribution including renal lesions. CASE PRESENTATION: A 51-year-old Japanese female was referred because of bilateral parotid swelling and renal dysfunction. Computed tomography scan showed the swelling of bilateral kidneys, parotid glands, and uterus. Ga scintigraphy also showed remarkable accumulation in these organs. Renal biopsy and cytological evaluations of parotid gland and uterus were performed and she was diagnosed as sarcoidosis of these organs. Treatment was initiated with prednisolone 40 mg/day and then renal dysfunction subsequently improved. In addition, the swelling of parotid glands and uterus improved and Ga accumulation in each organ had disappeared. CONCLUSION: This is a first case of renal sarcoidosis complicated by parotid glands and uterus lesions. Pathological findings and the reactivity observed in Ga scintigraphy indicated the presence of lesions in these organs.

Sialographic Analysis of Radioiodine-Associated Chronic Sialadenitis.

OBJECTIVES/HYPOTHESIS: To apply a novel sialography classification system to identify parotid and submandibular ductal findings following I-131 therapy and to assess correlates to dose and duration of symptoms. STUDY DESIGN: Retrospective single-center case series. METHODS: Patients who underwent sialography between February 2008 and February 2019 after previously receiving I-131 treatment were identified via a retrospective chart review. Their sialograms were systematically evaluated and scored by applying the Iowa parotid sialogram scale to also include submandibular gland analysis. RESULTS: From 337 sialograms, 30 (five submandibular, 25 parotid) underwent analysis. Ductal stenosis was identified in all sialograms and was graded as moderate (>50%-75%) in 7/30 cases and severe (>75%) in 15/30 cases. The distal (main) duct was narrowed in 23/30 cases. No association was identified between degree of ductal stenosis and I-131 dose (P = .39), age (P = .81), or time from I-131 therapy to sialogram (P = .97). CONCLUSIONS: The Iowa parotid sialogram scale was successfully applied to report abnormalities of the parotid and submandibular ductal system. The most common manifestation of I-131-associated sialadenitis was a severe stenosis within the distal salivary duct. No statistically significant association was found between degree of ductal stenosis and dose of I-131, age, or duration of symptoms. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E1450-E1456, 2021.

Differences between planned and delivered dose for head and neck cancer, and their consequences for normal tissue complication probability and treatment adaptation.

BACKGROUND AND PURPOSE: Anatomical changes induce differences between planned and delivered dose. Adaptive radiotherapy (ART) may reduce these differences but the optimal implementation is insufficiently clear. The aims of this study were to quantify the difference between planned and delivered dose in HNC patients, assess the consequential difference in normal tissue complication probability (ΔNTCP) and to explore the value of ΔNTCP as an objective selection strategy for ART. MATERIALS AND METHODS: For 52 patients, daily doses were accumulated to estimate the delivered dose. The difference from planned dose was analyzed for CTVs and 9 organs-at-risk (OAR). ΔNTCP was calculated for xerostomia, dysphagia, parotid gland dysfunction and tube feeding dependency at 6 months. ART was deemed necessary if ΔNTCP was >5%. The positive predicted value (PPV) was calculated for identification of ART-patients by clinical judgement, and ΔNTCP at fraction 10 and 15. RESULTS: ΔNTCP >5% was seen five times for dysphagia and twice for the other toxicities. Only 5/9 patients with any ΔNTCP >5% clinically received ART, although ART had been done for 13/52 patients (PPV: 0.38). PPV was 0.86 and 0.75 for accumulated dose at fraction 10 and 15, respectively, using a ΔNTCP cut-off for the allocation of ART of 5%. Using other ΔNTCP cut-offs did not substantially improve PPV. With this cut-off the negative predictive value was 0.93 for ΔNTCP method of fraction 10 and fraction 15, and 0.90 for clinical judgement. CONCLUSION: To identify patients accurately for ART, NTCP calculations based on the dose differences between planned and delivered dose at fraction 10 are superior to clinical judgement.

Bilateral multiple oncocytic cysts of the parotid gland in type 2 diabetes patients.

AIMS: The hallmarks of type 2 diabetes (T2D) are hyperglycaemia and insulin resistance. These factors, at the cellular level, are associated with mitochondrial dysfunction and increased glucose uptake. Such events are poorly explored in the context of the salivary glands. In this study, we present a series of eight cases of a distinct salivary gland lesion characterised by multiple oncocytic cysts, and we provide new pathological insights regarding its pathogenesis. METHODS AND RESULTS: Seven patients (87.5%) had confirmed T2D, and obesity was identified in five (62.5%) patients. Clinically, the patients showed bilateral parotid gland swelling with recurrent episodes of pain and enlargement. Imaging examination revealed multiple cystic lesions in both parotid glands. Microscopically, the parotid glands showed multiple cysts of different sizes, lined by oncocytic epithelial cells. Intraluminally, strongly eosinophilic glass-like crystalloid material was observed. Immunohistochemical studies were performed, and the most notable finding was glucose transporter 1 (GLUT1) overexpression in the oncocytic cysts which is not observed in any other oncocytic lesion of patients without T2D. In addition, high expressions of mitochondrial antigen, fission 1 protein and mitofusin-2 were observed in the oncocytic epithelium of the cysts. Furthermore, most of the oncocytic cysts showed a pattern of cytokeratin expression consistent with striated ducts. CONCLUSIONS: These results strongly suggest that T2D is associated with alterations in GLUT1 expression in the cells of striated ducts with mitochondrial dysfunction, causing a hyperplastic process characterised by multiple oncocytic cysts. For this lesion, the designation of 'diabetes-associated-bilateral multiple oncocytic cysts of the parotid gland' is proposed.

A Functional Approach to Posttraumatic Salivary Fistula Treatment: The Use of Botulinum Toxin.

This manuscript highlights key aspects regarding the practical use of botulinum toxin for the conservative nonsurgical treatment of a rarely encountered, but significant posttraumatic complication-the parotid salivary fistula. It adds information to the scarce existing literature on the subject. The authors outline the main differences between postoperative and trauma-related parotid injury regarding salivary fistula treatment. A total of 6 patients with trauma-related salivary fistulas have been treated by Abobotulinum toxin A injections over the course of 5 years. The technique is detailed, describing the doses used in the presence of parenchyma and duct injuries, the location and number of injection points in relation to the wound pattern. The results were favorable, leading to the healing of the salivary fistulas in all patients, with 1 injection session, without additional conservative treatment. In our experience, the use of botulinum toxin is of great benefit for treating salivary fistulas in a traumatic context.

Mumps presenting with unilateral, synchronous parotid and submandibular gland swelling.

A previously healthy 22-year-old woman presented with acute, unilateral facial and neck swelling, associated with fever and malaise. She was initially treated with intravenous antibiotics; however, CT imaging showed unilateral, synchronous swelling and inflammation of the parotid and submandibular glands, and a PCR swab from the parotid duct was positive for mumps. She was fully immunised and had no contact in the preceding period with anyone diagnosed with mumps. She responded to supportive management and her symptoms resolved over the course of her admission. Unilateral, synchronous swelling and severe inflammation of both the parotid and submandibular glands in mumps is a very unusual presentation, and not one previously reported in the literature.

Eosinophilic infiltrate resembling eosinophilic cellulitis (Wells syndrome) in a patient with mycosis fungoides.

Mycosis fungoides (MF) is a T-cell, non-Hodgkin lymphoma that primarily involves the skin. Extracutaneous involvement, such as in the parotidgland, is characteristic of end-stage disease. Eosinophilic cellulitis, or Wells syndrome, is a rare inflammatory dermatitis that involves a dermal infiltrate of eosinophils. We report a case of an 80-year-old man with a long-standing diagnosis of stage IIB MF who acutely developed parotid gland involvement and marked hypereosinophilia that most likely represented eosinophilic cellulitis. Activated T cells from his MF were likely a trigger factor for the development of his eosinophilic cellulitis. To our knowledge, this is the first reported case of an MF patient with atypical parotid gland involvement andeosinophilic cellulitis.

Cystinuria: One Possible Reason for Kidney and Salivary Gland Lithiasis Relationship.

Salivary gland lithiasis affects 1 to 2% of adults. The submandibular glands are concerned in 87% of cases. An association between kidney and salivary lithiases, although often mentioned, has rarely been observed. We relate an exceptional case of parotid and submandibular cystine sialolithiasis with kidney cystine lithiasis in a patient with cystinuria. Cystine lithiasis occurs in 1% of kidney lithiasis cases. The purpose of this article is to discuss the arguments, diagnosis, and therapeutic attitude in front of salivary cystine lithiasis. The patient was a 56-year-old woman treated for cystinuria undergoing a consultation for parotid lithiases. We obtained a panoramic view and performed a cervicofacial computed tomography scan, sialo-magnetic resonance imaging, and sialendoscopy. We found multiple lithiases in the distal portions of the Stensen duct, and 2 days after sialendoscopy, the patient expelled small sialoliths. Salivary lithiasis in patients with cystinuria has the same biochemical composition as kidney lithiasis. A computed tomography scan is efficient in most cases, but sialo-magnetic resonance imaging may be the only noninvasive method to diagnose small cystine salivary lithiases, such as those that can be seen in patients with cystinuria. Sialendoscopy is still an efficient diagnostic and therapeutic method for every type of salivary lithiasis.

Lipoedematous scalp: is there an association with fatty infiltration of the parotid?

Lipoedematous scalp (LS) is an extremely rare condition characterised by a soft and boggy consistency in the scalp due to an increased layer of subcutaneous tissue.In this report, we present a case of LS in a 64-year-old Indian woman. Clinical examination revealed only vague boggy lumpiness involving the whole of occipital scalp extending to parietal scalp. MRI scalp showed diffuse fatty infiltration of the scalp, particularly at the posterior parietal and occipital convexity extending to both lateral aspects of the cranium, with homogeneous signal in keeping with fat. Incidentally MRI also found diffuse fatty infiltration of the parotids.The aethiopathogenesis of LS is still unknown, however it is believed that the hormone leptin could be the key hormone in the dysregulation of fat deposition and distribution. This case report highlights the subtle features with which these cases can present and explores the literature on reported cases of LS.

Human immunodeficiency virus-associated cystic lymphoid hyperplasia: An immunohistochemical description.

BACKGROUND: Cystic lymphoid hyperplasia (CLH) describes benign salivary lymphoepithelial cysts with a strong link to human immunodeficiency virus (HIV) infection and acquired immunodeficiency syndrome (AIDS). The pathogenesis is related to ductal dilatation of entrapped salivary elements due to intranodal HIV-associated atypical lymphoid hyperplasia. Very little is known about the immunophenotypic profile of this entity. AIM: This study aims to describe the immunopathological features of a series of CLH cases in HIV-positive patients to clarify the etiopathogenesis. MATERIALS AND METHODS: Paraffin-embedded tissue from 25 cases of parotid CLH in HIV seropositive patients was immunohistochemically analyzed with CD3, CD20, CD4, CD8, and p24 using standard procedures. STATISTICAL ANALYSIS: The data are mostly descriptive and were analyzed using EpiInfo (3.5.1) (CDC, Atlanta, USA); significant differences were analyzed using the Student's t-test and the Chi-square test with a statistical significance level of P < 0.05 being used. RESULTS: Immunostaining showed a CD8:CD4 of ~1:1 except in selected cases with decreased CD4 and increased CD8 expression in the interfollicular (IF) areas. p24 staining revealed 100% specificity in HIV-associated CLH. CONCLUSION: The immunohistochemical description of CD20, CD3, CD4, and CD8 provides an understanding of CLH pathogenesis. CLH of parotid lymph nodes in confirmed HIV-positive patients with 100% specificity with HIV p24 antibody validates the strong association of CLH with HIV and AIDS. The CD4:CD8 ratio was ~1:1; however, increased CD8 expression within IF areas may indicate possible HIV-related CLH as compared to other cystic parotid lesions.

[Calcifications of the parotid space. A review]. / Calcifications de la loge parotidienne. Mise au point.

INTRODUCTION: Parotid lithiasis is the main cause of calcifications in the parotid space. However, there are many other less known causes. The aim of our study was to point out the non-lithiasic causes of calcifications in the parotid space. MATERIAL AND METHODS: We conducted an exhaustive review of the literature by mean of PubMed, using the keywords "parotid" and "calcification" and limiting our analysis to the original articles in humans published in English and in French. Articles reporting about microscopic calcifications and who were not dealing with parotid calcifications were excluded. RESULTS: Twenty articles met the inclusion criterions. Tumoral and non-tumoral local causes and systemic causes of parotid calcification were found. The way they revealed was variable. The main tumoral local causes were pleomorphic adenomas, salivary duct carcinomas and adenocarcinomas. The main non-tumoral local causes included vascular malformations and calcified parotid lymph nodes. The main systemic causes were chronic kidney diseases, HIV infection, chronic alcoholism, elevated levels of alkaline phosphatase and auto-immune diseases. DISCUSSION: Eighteen different etiologies of parotid space calcifications could be identified. First line exploration of these lesions relies mainly on conventional radiography and ultrasound examination that are easily available. CT scan remains the reference examination.

Necrotizing sialometaplasia of the parotid gland associated with facial nerve paralysis.

INTRODUCTION: Necrotizing sialometaplasia is a benign inflammatory lesion involving most frequently the minor salivary gland of the hard palate. Involvement of the parotid gland is rare, involvement of the parotid gland associated with facial palsy is exceptional. CASE REPORT: A 56-year-old male patient with Marfan syndrome presented with swelling and inflammation of the left parotid gland associated with progressively complete facial nerve paralysis. CT scan and MRI showed a parotid collection with hyper signal of the nearest tissues associated with erosion of the styloid process. A malignant tumor was suspected. The histological examination of a biopsy showed a lobulocentric process with necrosis, squamous metaplasia, and inflammation. The immunohistochemical examination supported a final diagnosis of necrotizing sialometaplasia. DISCUSSION: Necrotizing sialometaplasia of the parotid gland associated with facial nerve paralysis presents like a malignant neoplasm, both clinically and histologically. Only advanced immunohistochemical examination can really confirm the diagnosis.

Plunging ranula with prestyloid parapharyngeal space, masticator space, and parotid gland extension.

Plunging ranula with prestyloid parapharyngeal space, masticator space, and parotid gland extension. INTRODUCTION: Ranulas develop from mucous extravasation secondary to sublingual gland duct obstruction or trauma. Plunging ranula usually dive into the submandibular space. METHODS: This is the first reported case of a plunging ranula with direct extension to the prestyloid parapharyngeal space, masticator space, and parotid gland with avoidance of the submandibular space. RESULTS: The patient presented with a tender parotid mass, of which the differential is broad, including parotitis, parotid malignancy, metastatic malignancy, lymphoma, as well as other infectious etiologies. When an intraoral component is not identified, other differential considerations would be thyroglossal duct cyst, branchial cleft cyst, parathyroid cyst, cervical thymic cyst, dermoid cyst, cystic hygroma, or benign teratoma. CONCLUSION: The case is unique due to ranula extension into multiple spaces. For optimal treatment, the sublingual gland along with its tract and contents needs to completely removed.

Surgical management of parotid salivary duct rupture secondary to non-iatrogenic trauma in a dog.

A 3-year-old French bulldog presented for evaluation of recurrent swelling and a fistula on the right cheek after a dog fight. A large volume of serous fluids was identified on the wound immediately after atropine drops. A diagnosis of parotid salivary duct rupture secondary to trauma was made. On surgical exploration, the thickened proximal segment of the severed duct was identified and circumferentially double ligated with 3-0 silk. No evidence of swelling and normal appearance of the parotid salivary gland were identified 4 months postoperative recheck. No further problems were noted 10 months postoperatively phone-call. To the author's knowledge, this is the first reported case of successful proximal parotid duct ligation of parotid salivary duct rupture secondary to non-iatrogenic trauma.