Escisión de Glándula Submandibular por Sialoadenitis Crónica Esclerosante Asociada a Sialolito en el Conducto Submandibular / Submandibular Gland Excision Due to Chronic Sclerosing Sialadenitis Associated With Sialolith in Submandibular Duct

La sialoadenitis crónica esclerosante puede extenderse desde una sialoadenitis focal hasta una cirrosis completa de la glándula. Aparece entre los 40 y 70 años de edad y afecta principalmente a la glándula submandibular. Se asocia con sialolitos y agentes infecciosos inespecíficos. La causa más frecuente de sialolitiasis es la formación de cálculos macroscópicamente visibles en una glándula salival o en su conducto excretor, de los cuales el 80 % al 90 % provienen de la glándula submandibular. Esta predilección probablemente se deba a que su conducto excretor es más largo, más ancho y tiene un ángulo casi vertical contra la gravedad, contribuyendo así a la estasis salival. Además, la secreción semimucosa de la misma es más viscosa. El sitio principal de ubicación de los litos en el conducto submandibular es la región hiliar con un 57 %. La sintomatología típica de la sialolitiasis es el cólico con tumefacción de la glándula y los dolores posprandiales. Reportamos el caso de un paciente masculino de 55 años, quien ingresa al servicio de Cirugía Oral y Maxilofacial del Hospital General Balbuena de la Ciudad de México por presentar un aumento de volumen en la región submandibular izquierda de consistencia dura y dolorosa a la palpación de 15 días de evolución, acompañada de aumento de temperatura en la zona compatible con un absceso. Los estudios de imagen reportan un sialolito en la región hiliar del conducto submandibular de 2,0 x 1,7 x 1,0 cm. Debido a su localización y tamaño, el tratamiento ideal en estos casos es la escisión de la glándula junto con el lito previo drenaje del absceso e inicio de terapia antibiótica doble.

Chronic sclerosing sialadenitis can range from focal sialadenitis to complete cirrhosis of the gland. It appears between 40 and 70 years of age and mainly affects the submandibular gland. It is associated with sialoliths and nonspecific infectious agents. The most common cause of sialolithiasis is the formation of macroscopically visible stones in a salivary gland or its excretory duct, of which 80 % to 90 % come from the submandibular gland. This predilection isprobably due to the fact that their excretory duct is longer, wider and has an almost vertical angle against gravity, thus contributing to salivary stasis. In addition, the semimucous secretion of it is more viscous. The main location of the stones in the submandibular duct is the hilar region with 57 %. The typical symptomatology of sialolithiasis is colic with swelling of the gland and postprandial pain. We report the case of a 55-year-old male patient, who was admitted to the Oral and Maxillofacial Surgery Service of the Hospital General Balbuena in Mexico City due to an increase in volumen in the left submandibular region that was hard and painful on palpation of 15 days of evolution, accompanied by increased temperature in the area compatible with an abscess. Imaging studies report a 2.0 x 1.7 x 1.0 cm sialolith in the submandibular duct hilar region. Due to its location and size, the ideal treatment in these cases is excision of the gland together with the stone previous drainage of the abscess and initiation of dual antibiotic therapy.

Evaluation of immune microenvironment in IgG4-related sialadenitis.

IgG4-related sialadenitis is a systemic autoimmune disease that can lead to fibro-inflammatory conditions. This study aims to investigate the immune microenvironment and potential signaling pathways associated with IgG4-related sialadenitis. Datasets related to IgG4-related sialadenitis were retrieved from the GEO database. Immune cell infiltration analysis was conducted using the Cell-type Identification by Estimating Relative Subsets of RNA Transcripts (CIBERSORT) method. Differentially immune-related expressed genes (DIEG) and immune-related functional enrichment were identified. Moreover, potential treatment targets for IgG4-related sialadenitis were predicted using The Connectivity Map. Only two datasets from GEO were included for further analysis. The CIBERSORT results indicated dominant immune cell populations in IgG4-related sialadenitis, including CD8+ T cells, resting NK cells, monocytes, and naïve B cells in peripheral blood mononuclear cells. Additionally, high abundance of plasma cells was observed in labial salivary gland tissues. Furthermore, a total of 42 DIEGs were identified, with tumor necrosis factor (TNF) signaling via the NF-Kappa B signaling pathway and the p53 signaling pathway being highly enriched. Autophagy inhibitors and DNA topoisomerase inhibitors were strongly associated with potential targets for the treatment of IgG4-related sialadenitis (P<0.05). This study reveals altered immune microenvironment in peripheral blood mononuclear cells and labial salivary gland tissues in IgG4-related sialadenitis. Autophagy inhibitors and DNA topoisomerase inhibitors show promise as potential targets for treating IgG4-related sialadenitis, providing a novel therapeutic strategy for this disease.

A conservative treatment for chronic obstructive sialoadenitis by intraductal instillation of mucolytic, steroids and antibiotic solution.

PURPOSES: Reporting our experience in treating chronic obstructive sialadenitis with a protocol consisting of sialoendoscopy and intraductal instillation of antibiotics, steroids and n-acetyl-cysteine (NAC) solution. METHODS: Prospective study of patients with chronic obstructive sialadenitis with no apparent lithiasic obstructions, with recurrent non-lithiasic sialoadenitis and patients with lithiasic sialoadenitis not solved with sialoendoscopy. In all cases, a sialoendoscopy was performed. All the patients affected by lithiasic sialoadenitis where the chronic inflammation was resolved with sialoendoscopy were excluded from the study. The mid-term follow-up was performed at 12 months via phone interview, to understand whether patients had developed any further symptoms after the treatment. RESULTS: This study included 26 patients. All the patient without sialolithiasis have not reported any symptoms during the follow-up period. Two of those with sialolithiasis have not shown any signs of recurrence. The remaining three patients with non-resolved sialolithiasis had a recurrence of symptoms which were treated again with 1 intraductal administration of betamethasone, gentamicine and NAC, showing immediately a regression of the symptoms. CONCLUSIONS: Intraductal administration of gentamicin + NAC + betamethasone seemed effective for the therapy of chronic obstructive sialoadenitis. Our protocol seemed effective also in that cases where it was not possible to remove or detect endoscopically an obstruction. In all these cases we have noticed an increase in the symptom-free time even in cases where it was not possible to remove the stones.

Suspected IgG4-related disease of the submandibular salivary gland and periorbital soft tissue in a man with latent tuberculosis.

A 77-year-old Lithuanian man presented to our institution with recurrent episodes of periorbital cellulitis, submandibular swelling and sialadenitis. Investigations revealed a positive QuantiFERON, raised inflammatory markers and normal autoimmune screen. Cross-sectional imaging showed no signs of occult malignancy, and work-up for mycobacterial infection including imaging and bronchoalveolar lavage did not show active tuberculosis. During hospitalisation, the patient developed fevers of unknown origin, which were investigated with a positron emission tomography (PET) scan and a bone marrow aspiration, without evidence of occult infection or malignancy. Serum IgG4 level was three times the upper limit of normal. The patient responded well to oral steroids but relapsed after completing a slow taper. Serum IgG4 level was three times the upper limit of normal. He had an American College of Rheumatology/European League Against Rheumatism score of 20, in conjunction with involvement of orbital and salivary tissue. Therefore, IgG4-related disease was considered the most likely diagnosis, despite prominent fevers, which are among the exclusion criteria for this diagnosis. After a multidisciplinary review including rheumatology and ophthalmology, the patient was commenced on maintenance methotrexate with remission of symptoms.

Methicillin-resistant Staphylococcus aureus (MRSA) submandibular abscess in a neonate.

Neonatal suppurative submandibular sialadenitis and abscess formation is an exceedingly rare entity. This report describes a complex case of a male neonate with a methicillin-resistant Staphylococcus aureus (MRSA) submandibular abscess, requiring emergency intubation due to acute airway compromise. The patient was admitted to the paediatric intensive care unit, received appropriate antibiotic treatment and underwent urgent surgical drainage of the abscess. He made a full recovery and remains well 18 months later. No comorbidities or common risk factors for the disease were identified. Although extremely uncommon, neonatal submandibular abscesses can lead to significant morbidity. Neonates tend to present insidiously, and sudden clinical deterioration with airway compromise is possible. MRSA has been increasingly implicated in these infections, even in the absence of relevant risk factors. As such, continued clinical vigilance is essential for prompt diagnosis and prevention of life-threatening complications. Multidisciplinary input is paramount for appropriate management of these complex infections.

Clinical presentation and microbiology of acute salivary gland infections.

INTRODUCTION: Acute salivary gland infections (ASI) have been associated with poor outcome in elderly and postoperative patients. Perioperative care and treatment of co-morbidities have improved considerably, but most of our knowledge regarding ASI dates back several decades. The aim of this study was to describe the microbiology and treatment of ASI in a large post-millennial cohort. METHODS: All patients with ASI admitted to the Department of Otorhinolaryngology - Head and Neck Surgery, Aarhus University Hospital in the period from 2001 to 2017 were included. RESULTS: In total, 157 patients with ASI were included. The parotid gland (PG) was affected in 89 (57%) cases and the submandibular gland (SMG) in 68 (43%) cases. The most prevalent bacterial findings were viridans streptococci (25 isolates) and Staphylococcus aureus (19 isolates). S. aureus was almost exclusively found in PG (17/19 cases). S. aureus-positive cases showed a significantly higher inflammatory response than other bacteria (C-reactive protein, p = 0.008 and absolute neutrophil count, p = 0.0108). CONCLUSIONS: S. aureus is a significant pathogen in ASI and especially in PG cases. Other pathogens may play a role in the development of SMG infections. Based on the bacterial findings in this study, we recommend penicillinase-resistant penicillin as first-line treatment in ASI. FUNDING: none Trial registration: not relevant. The Danish Data Protection Agency approved the project.

Transgenic rice seeds expressing altered peptide ligands against the M3 muscarinic acetylcholine receptor suppress experimental sialadenitis-like Sjögren's syndrome.

Objective: We previously reported that Rag1-/- mice inoculated with splenocytes from M3 muscarinic acetylcholine receptor (M3R) knockout mice immunized with an M3R peptide mixture developed sialadenitis-like Sjögren's syndrome (M3R-induced sialadenitis [MIS]). We also found that intravenous administration of altered peptide ligand (APL) of N-terminal 1 (N1), which is one of the T-cell epitopes of M3R, suppressed MIS. In this study, we aimed to evaluate the suppressive ability and its mechanisms of rice seeds expressing N1-APL7 against MIS.Methods: Rice seeds expressing N1 and N1-APL7 were orally administered to MIS mice for 2 weeks. The changes in saliva flow and sialadenitis (salivary gland inflammation) were analyzed. The M3R-specific T-cell response in the spleen and the expression of regulatory molecules in the cervical lymph nodes and mesenteric lymph nodes were also analyzed.Results: Oral administration of N1-APL7-expressing rice seeds significantly recovered reduction in saliva flow and suppressed sialadenitis when compared with treatment with nontransgenic rice seeds and N1 rice seeds. IFNγ production from M3R-reactive T cells tended to decline in the N1-APL7 rice-treated group as compared with those in the other groups. In the N1-APL7 rice-treated group, the mRNA expression levels of Foxp3 in the cervical-lymph-node CD4+ T cells were higher than those in the other groups.Conclusion: Oral administration of N1-APL7-expressing rice suppressed MIS via suppression of M3R-specific IFNγ and IL-17 production and via enhancement of regulatory molecule expression.Key messagesWe generated N1-peptide- or N1-APL7-expressing rice seeds. Oral administration of N1-APL7-expressing rice seeds significantly recovered the reduction of saliva flow and suppressed sialadenitis via the suppression of M3R specific IFNγ and IL-17 production and via enhancement of regulatory T (Treg) cells.

Blockade of CD40-CD154 pathway interactions suppresses ectopic lymphoid structures and inhibits pathology in the NOD/ShiLtJ mouse model of Sjögren's syndrome.

OBJECTIVE: To examine the role of CD40-CD154 costimulation and effects of therapeutic pathway blockade in the non-obese diabetic (NOD/ShiLtJ) model of Sjögren's syndrome (SS). METHODS: We assessed leucocyte infiltration in salivary glands (SGs) from NOD/ShiLtJ mice by immunohistochemistry and examined transcriptomics data of SG tissue from these animals for evidence of a CD40 pathway gene signature. Additionally, we dosed MR1 (anti-CD154 antibody) in NOD mice after the onset of SS-like disease and examined the effects of MR1 treatment on sialadenitis, autoantibody production, SG leucocyte infiltration, gene expression downstream of CD40 and acquaporin 5 (AQP5) expression. RESULTS: We could detect evidence of CD40 expression and pathway activation in SG tissue from NOD mice. Additionally, therapeutic treatment with MR1 suppressed CD40 pathway genes and sialadenitis, inhibited ectopic lymphoid structure formation and autoantibody production, as well as decreased the frequency of antibody-secreting cells in SGs but had minimal effects on AQP5 expression in NOD/ShiLtJ SGs. CONCLUSION: CD40-CD154 interactions play an important role in key pathological processes in a mouse model of SS, suggesting that blockade of this costimulatory pathway in the clinic may have beneficial therapeutic effects in patients suffering from this autoimmune exocrinopathy.

Phosphatidylinositol 3-kinase delta pathway: a novel therapeutic target for Sjögren's syndrome.

BACKGROUND: The phosphatidylinositol 3-kinase delta isoform (PI3Kδ) belongs to an intracellular lipid kinase family that regulate lymphocyte metabolism, survival, proliferation, apoptosis and migration and has been successfully targeted in B-cell malignancies. Primary Sjögren's syndrome (pSS) is a chronic immune-mediated inflammatory disease characterised by exocrine gland lymphocytic infiltration and B-cell hyperactivation which results in systemic manifestations, autoantibody production and loss of glandular function. Given the central role of B cells in pSS pathogenesis, we investigated PI3Kδ pathway activation in pSS and the functional consequences of blocking PI3Kδ in a murine model of focal sialoadenitis that mimics some features of pSS. METHODS AND RESULTS: Target validation assays showed significant expression of phosphorylated ribosomal protein S6 (pS6), a downstream mediator of the phosphatidylinositol 3-kinase delta (PI3Kδ) pathway, within pSS salivary glands. pS6 distribution was found to co-localise with T/B cell markers within pSS aggregates and the CD138+ plasma cells infiltrating the glands. In vivo blockade of PI3Kδ activity with seletalisib, a PI3Kδ-selective inhibitor, in a murine model of focal sialoadenitis decreased accumulation of lymphocytes and plasma cells within the glands of treated mice in the prophylactic and therapeutic regimes. Additionally, production of lymphoid chemokines and cytokines associated with ectopic lymphoneogenesis and, remarkably, saliva flow and autoantibody production, were significantly affected by treatment with seletalisib. CONCLUSION: These data demonstrate activation of PI3Kδ pathway within the glands of patients with pSS and its contribution to disease pathogenesis in a model of disease, supporting the exploration of the therapeutic potential of PI3Kδ pathway inhibition in this condition.

Poor patient compliance with instructions for continuous sialogogues after 131 I therapy.

OBJECTIVES: To analyze the role of patient compliance as a factor in evaluating the effectiveness of continuous sialogogues to prevent salivary side effects from 131 I therapy in differentiated thyroid cancer patients. METHODS: Differentiated thyroid cancer patients who were clinically scheduled for an 131 I therapy at MedStar Washington Hospital Center between 2012 and 2013 were given instructions for continuous sialogogues per standard clinical protocol. The prospective survey was given at multiple time points. RESULTS: Ninety-nine patients consented to participate of whom 94 participants had complete data. The mean prescribed 131 I activity was 121 ± 50 mCi (4.5 ± 1.9 GBq), range 27.5-288 mCi (1.0-10.7 GBq ). Overall, only 10% (9/94) of patients were compliant with continuous sialogogues. Even though all patients took sialogogues on the first day of post-therapy, 17% of participants did not continuously take sialogogues during the first day, 60% during the first night, and 72% on the second day despite rigorous instructions to continue for two days. CONCLUSION: Despite repetitive instructions to use sialogogues continuously, most patients (90%) were not compliant. In future studies, strict monitoring and evaluation of patient compliance will be crucial when assessing the effect of continuous versus intermittent or delayed initiation of sialogogues.

Incidence of Acute Suppurative Sialadenitis in End-Stage Cancer Patients: A Retrospective Observational Study.

CONTEXT: Acute suppurative sialadenitis (hereafter referred to as sialadenitis) is accompanied by pain and fever and can diminish the quality of life in end-stage cancer patients; however, its incidence is not clear. OBJECTIVES: We conducted this study to elucidate the incidence of sialadenitis in end-stage cancer patients. METHODS: Retrospective review and observational study based on patients' medical records. SUBJECTS: About 726 consecutive cancer patients who died on the palliative care unit of our hospital between April 2012 and November 2016 were included. MEASUREMENTS: Median duration between sialadenitis onset and death, concomitant treatment, average infusion volume per day, site of onset, symptoms, effectiveness of antibiotic treatment, and mean duration until symptomatic relief. RESULTS: The incidence of sialadenitis was 2.9% (21 of 726 cases). The median duration from onset to death was 20 days (range 2-112); concomitant treatment included opioids in 11 patients (55%), anticholinergic drugs in six patients (28%), steroids in three patients (14%), and oxygen inhalation in five patients (23%); average infusion volume per day was 588 ± 307 mL; site of onset was submandibular gland in 12 patients (57%) and parotid gland in nine patients (42%); and symptoms were pain in 18 patients (85%) and fever in 13 patients (61%). Antibiotic treatment was administered in 18 patients (85%), and the mean duration until symptomatic relief was 4.0 ± 1.5 days. CONCLUSION: Sialadenitis is a rare complication in end-stage cancer patients; however, it is important to recognize that it can be associated with severe symptoms, including fever and pain.

Combined interventional sialendoscopy and intraductal steroid therapy for recurrent sialadenitis in Sjögren's syndrome: Results of a pilot monocentric trial.

OBJECTIVES: To evaluate the effectiveness of interventional sialendoscopy alone or combined with outpatient intraductal steroid irrigations in patients with sialadenitis due to Sjögren's syndrome (SS). DESIGN: A pilot therapeutic study. SETTING: ENT Clinics, Universities of Milan and Pavia. STUDY POPULATION: We included 22 patients with SS of whom 12 underwent interventional sialendoscopy followed by intraductal steroid irrigations (group A), and 10 interventional sialendoscopy alone (group B). OUTCOMES MEASURES: The following outcome measures were considered and recorded before and after the therapeutic intervention: (i) number of episodes of glandular swelling, (ii) cumulative prevalence of patients with glandular swelling assessed by the specific domain, the EULAR SS Disease Activity Index (ESSDAI), (iii) severity of pain by means of a 0-10 pain visual analogue scale (VAS), (iv) severity of xerostomia and other disease symptoms assessed by the EULAR SS Patient Reported Index (ESSPRI) and the Xerostomia Inventory questionnaire. RESULTS: The postoperative reduction in the mean number of episodes of glandular swelling was 87% (95% CI: 77-93) and 75% (95% CI: 47%-88%) in the groups A and B, respectively. The percentage of patients with glandular swelling decreased from 41.7% to 0.0% in the group A and from 30.0% to 0.0% in the group B, respectively. Most of the patients experienced a subjective clinical improvement documented by the statistically significant reductions in the postoperative mean pain VAS (group A P<.001; group B P=.004), Xerostomia Inventory (P<.001 and P=.003) and ESSPRI scores (P<.001 and P=.008). Interventional sialendoscopy followed by outpatient intraductal steroid irrigations was more effective than interventional sialendoscopy alone, when pain VAS, Xerostomia Inventory and ESSPRI scores before and after treatment were analysed together using the multivariate Hotelling T2 test (P=.0173). CONCLUSIONS: This pilot study confirms that interventional sialendoscopy with steroid duct irrigation significantly reduces the number of painful episodes of sialadenitis and improves the subjective sensation of oral dryness and other disease symptoms in patients with SS. The study results also suggest that the improvement is greater when interventional sialendoscopy is combined with a cycle of outpatient steroid ductal irrigations. Larger controlled randomised studies are certainly needed to confirm these preliminary data.

Recent advances in knowledge regarding the head and neck manifestations of IgG4-related disease.

IgG4-related disease (IgG4-RD) is a chronic inflammatory disorder, characterized by elevated serum IgG4 levels as well as abundant infiltration of IgG4-positive plasmacytes and fibrosis in various organs, including the head and neck region. In particular, the salivary glands, orbit, and thyroid are common sites of disease involvement. IgG4-RD is diagnosed based on various clinical, serological, and histopathological findings, none of which are pathognomonic. Hence, various differential diagnoses, which exhibit elevated serum IgG4 levels and infiltration of IgG4-postive cells into tissues, need to be excluded, especially malignant diseases and mimicking disorders. Systemic corticosteroids are generally effective in inducing IgG4-RD remission; however, recurrent or refractory cases are common. In addition, although the pathogenic mechanisms of IgG4-RD remain unclear, an antigen-driven inflammatory condition is believed to be involved. Recent studies have indicated the important pathogenic role of B cell/T cell collaboration and innate immunity in this disease. Nevertheless, additional research and discussions are needed to resolve many remaining questions. In this review, we provide an overview of the recent insights on the history, clinical features, diagnosis, and treatment of IgG4-RD in the head and neck region. Furthermore, we have also addressed the pathogenesis of this disease.

Sclerotic effect of oxytetracycline on the submandibular gland: An experimental model.

Oxytetracycline has been suggested as an alternate therapy for chronic recurrent sialadenitis and sialorrhea. We conducted an experimental study to investigate the sclerotic effect of this drug on the submandibular gland by histopathologic methods. Our subjects were 20 New Zealand white rabbits, which were divided into two groups of 10. The right submandibular gland of the rabbits in the active-treatment group was injected with 0.3 ml of oxytetracycline (100 mg/ml), and that of the controls was injected with saline. Four weeks after the injections, all the glands were removed. Histopathologic studies, including hematoxylin and eosin and Masson trichrome staining, were carried out. The glands were evaluated for tissue inflammation, congestion, fibrosis, edema, lipomatosis, and atrophy. To investigate apoptosis, terminal deoxynucleotidyl transferase-mediated dUTP-digoxigenin nick-end labeling (TUNEL) immunohistochemical staining was used. In the study group, inflammation (n = 9), congestion (n = 9), fibrosis (n = 6), edema (n = 6), and lipomatosis (n = 4) were observed; in the sham group, only lipomatosis was seen (n = 5). The TUNEL assay results for acinar cells were 4.51 ± 1.41% in the oxytetracycline group and 2.08 ± 1.76% in the control group (p = 0.006); the corresponding figures for the duct cells were 7.05 ± 0.87% and 3.10 ± 2.26% (p = 0.001). Based on our findings, we conclude that oxytetracycline might be a viable alternative for the treatment of chronic recurrent sialadenitis and sialorrhea. However, more research in this area is needed.

Clinicopathology of Immunoglobulin G4-Related Chronic Sclerosing Sialadenitis: A Single-Center Study.

OBJECTIVES: To investigate the clinicopathologic characteristics of patients with immunoglobulin G4-related chronic sclerosing sialadenitis (IgG4-RCSS), a recently recognized disease. STUDY DESIGN: Case series with chart review and pathology study. SETTINGS: Tertiary care hospital. SUBJECTS AND METHODS: We evaluated chronic sialadenitis specimens obtained over 11 years using pathologic examination and IgG4 immunohistochemistry staining. The specimens were assigned a revised diagnosis of IgG4-RCSS or chronic sialadenitis not otherwise specified, and clinicopathologic data from each group were compared. RESULTS: Of the 84 patients, 21 were diagnosed with IgG4-RCSS and 63 with chronic sialadenitis not otherwise specified. IgG4-RCSS patients were older (68.2 ± 13.9 vs 54.2 ± 15.8 years, P = .001), predominantly male (85.7% vs 61.9%, P = .036), and more likely to present with painless swelling (75% vs 44.3%, P = .001) and bilateral involvement (52.4% vs 6.3%, P < .001). Ratio of IgG4-positive plasma cells to IgG-positive plasma cells in IgG4-RCSS tissues was 0.81 ± 0.14. The mean value of serum IgG4 in IgG4-RCSS patients was 918.8 mg/dL. CONCLUSION: IgG4-RCSS is more common in older male patients and frequently presents with bilateral involvement. Informing head and neck surgeons of the clinical features of IgG4-RCSS and promoting a combined approach of clinical evaluation, imaging, and biopsy can improve the accuracy of preoperative diagnoses.

Uncommon situation and presentation of chronic sclerosing sialadenitis.

Chronic sclerosing sialadenitis of the parotid gland is a very uncommon chronic inflammatory salivary gland disease. Clinically, it presents as a slow-growing painful. Histologically, it showed a chronic inflammation and fibrosis. This case report highlights the clinical, radiological and histological aspects of this disease. We report unusual case of chronic sclerosing sialadenitis of the parotid in a 12-year-old man. CT detected a mass of tissue density in the right parotid. The evolution was marked by spontaneous fistula allowing a surgical biopsy. The mass regressed after corticosteroids. The follow-up was normal. The location, age and presentation make our case very interesting.