A nomogram based on ultrasound scoring system for differentiating between immunoglobulin G4-related sialadenitis and primary Sjögren syndrome.

OBJECTIVES: Accurate distinguishing between immunoglobulin G4-related sialadenitis (IgG4-RS) and primary Sjögren syndrome (pSS) is crucial due to their different treatment approaches. This study aimed to construct and validate a nomogram based on the ultrasound (US) scoring system for the differentiation of IgG4-RS and pSS. METHODS: A total of 193 patients with a clinical diagnosis of IgG4-RS or pSS treated at our institution were enrolled in the training cohort (n = 135; IgG4-RS = 28, pSS = 107) and the validation cohort (n = 58; IgG4-RS = 15, pSS = 43). The least absolute shrinkage and selection operator regression algorithm was utilized to screen the most optimal clinical features and US scoring parameters. A model for the differential diagnosis of IgG4-RS or pSS was built using logistic regression and visualized as a nomogram. The performance levels of the nomogram model were evaluated and validated in both the training and validation cohorts. RESULTS: The nomogram incorporating clinical features and US scoring parameters showed better predictive value in differentiating IgG4-RS from pSS, with the area under the curves of 0.947 and 0.958 for the training cohort and the validation cohort, respectively. Decision curve analysis demonstrated that the nomogram was clinically useful. CONCLUSIONS: A nomogram based on the US scoring system showed favourable predictive efficacy in differentiating IgG4-RS from pSS. It has the potential to aid in clinical decision-making.

[Salivary gland infections or sialadenitis]. / Pathologies infectieuses des glandes salivaires ou sialadénites.

SALIVARY GLAND INFECTIONS OR SIALADENITIS. Salivary infections, or sialadenitis, affect all types of patients but are most common in elderly or dehydrated patients. Clinical signs include pain, skin erythema, gland oedema, fever and sometimes pus visible at the ostium. In the absence of doubt, no investigation is necessary apart from bacteriological sampling. Ultrasound scan should be performed if there is a doubt about the diagnosis or signs of complications. The treatment of bacterial sialadenitis is based on a probabilistic antibiotic therapy then adapted, for a duration of 10 days. The most frequent germ is Staphylococcus aureus. Rehydration is essential, especially in the elderly. The treatment of viral sialadenitis, which resolves in 8 to 15 days, is symptomatic. If the evolution is most often positive, the risk of serious complications requires a rapid diagnosis and appropriate treatment.

PATHOLOGIES INFECTIEUSES DES GLANDES SALIVAIRES OU SIALADÉNITES. Les infections salivaires, ou sialadénites, touchent tout type de patients mais concernent surtout des patients âgés ou déshydratés. Les signes cliniques incluent douleur, érythème cutané, oedème de la glande, fièvre et parfois pus visible à l'ostium. En l'absence de doute, aucun examen n'est utile en dehors des prélèvements. Une échographie est réalisée en cas d'incertitude diagnostique ou de signes de complications. Le traitement des sialadénites bactériennes repose sur une antibiothérapie probabiliste secondairement adaptée pour une durée de dix jours. Le germe le plus fréquent est Staphylococcus aureus. La réhydratation est primordiale, surtout chez les sujets âgés. Le traitement des sialadénites virales, résolutives en huit à quinze jours, est symptomatique. Si l'évolution est le plus souvent favorable, le risque de complications graves impose un diagnostic rapide et un traitement adapté.

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.

The Need for Studies on Oral Corticosteroids After Sialendoscopy for Obstructive Salivary Gland Disease: Systematic Review.

OBJECTIVES: This qualitative systematic review evaluates the evidence in support of the use of oral corticosteroids in patients undergoing sialendoscopy for the treatment of obstructive sialadenitis. DESIGN: Qualitative systematic review. METHODS: A literature search was conducted from January 1985 and September 2020. Inclusion criteria embraced peer-reviewed articles in which adult patients undergoing interventional sialendoscopy for obstructive salivary gland disease received oral corticosteroids. The results were initially screened based on title and abstract, and the remaining articles were reviewed for eligibility. RESULTS: About 218 papers were selected by title and abstract, 96 were selected for full-text review, and 9 met the inclusion criteria. Eight published reports were retrospective observational studies and 1 was a prospective comparative study. Overall, the heterogeneity of clinical data stood out in this systematic review. The pooled success rate in the studies was 873/979 (89%). Only 5 studies described a rationale for oral corticosteroid use as part of the post-operative management. In 4 studies, a prednisone total daily dose of 40 to 50 mg was used. One study clearly showed a lower recurrence rate in patients who received oral steroids for more than 7 days in addition to sialendoscopy for management of ductal stenoses. CONCLUSION: This systematic review showed that most centers that prescribe oral corticosteroids after sialendoscopy are unaware of the specific results with this treatment. For ductal stenoses, only 1 paper clearly showed the benefits of oral corticosteroids after sialendoscopy but more high-quality evidence is required in the form of a comparative study or randomized controlled trial, with appropriate long-term follow up.

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.

Cenerimod, a selective S1P1 receptor modulator, improves organ-specific disease outcomes in animal models of Sjögren's syndrome.

BACKGROUND: Sjögren's syndrome is a systemic autoimmune disease characterized by immune cells predominantly infiltrating the exocrine glands and frequently forming ectopic lymphoid structures. These structures drive a local functional immune response culminating in autoantibody production and tissue damage, associated with severe dryness of mucosal surfaces and salivary gland hypofunction. Cenerimod, a potent, selective and orally active sphingosine-1-phosphate receptor 1 modulator, inhibits the egress of lymphocytes into the circulation. Based on the mechanism of action of cenerimod, its efficacy was evaluated in two mouse models of Sjögren's syndrome. METHODS: Cenerimod was administered in two established models of Sjögren's syndrome; firstly, in an inducible acute viral sialadenitis model in C57BL/6 mice, and, secondly, in the spontaneous chronic sialadenitis MRL/lpr mouse model. The effects of cenerimod treatment were then evaluated by flow cytometry, immunohistochemistry, histopathology and immunoassays. Comparisons between groups were made using a Mann-Whitney test. RESULTS: In the viral sialadenitis model, cenerimod treatment reduced salivary gland immune infiltrates, leading to the disaggregation of ectopic lymphoid structures, reduced salivary gland inflammation and preserved organ function. In the MRL/lpr mouse model, cenerimod treatment decreased salivary gland inflammation and reduced T cells and proliferating plasma cells within salivary gland ectopic lymphoid structures, resulting in diminished disease-relevant autoantibodies within the salivary glands. CONCLUSIONS: Taken together, these results suggest that cenerimod can reduce the overall autoimmune response and improve clinical parameters in the salivary glands in models of Sjögren's syndrome and consequently may reduce histological and clinical parameters associated with the disease in patients.

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.

Pathogenic roles and therapeutic potential of the CCL8-CCR8 axis in a murine model of IgG4-related sialadenitis.

BACKGROUND: Our previous studies reveal that CCL18-CCR8 chemokine axis is upregulated in patients of immunoglobulin G4-related disease (IgG4-RD), suggesting that the CCL18-CCR8 axis is implicated in the etiology of IgG4-RD, although whether this axis has a potential as a therapeutic target remains unclear. Our purpose was to clarify the pathogenic roles and therapeutic potential of the murine CCL8 (analog of human CCL18)-CCR8 axis by using an animal model of IgG4-RD (LAT Y136F knockin mice; LAT mice). METHODS: We compared the infiltration of inflammatory cells and the fibrosis of the salivary glands of 6-week-old LAT mice and littermate mice. The expressions of Ccl8 and Ccr8 were also compared. Next, we investigated the therapeutic effects of intravenous administration of anti-CCL8 neutralizing antibody in LAT mice against inflammation and fibrosis of the salivary glands. We also investigated the effects of stimulation with recombinant mouse CCL8 on the collagen production in a mouse fibroblast cell line (NIH/3 T3) in vitro. RESULTS: When compared with the littermates, the LAT mice showed apparent infiltration of inflammatory cells and fibrosis in the salivary glands. The focus and fibrosis score in the salivary glands were significantly higher in the LAT mice than in the littermates. The expression levels of Ccl8 in the spleen and of Ccr8 in the salivary glands were significantly higher in the LAT mice than in the littermates. Anti-CCL8 antibody significantly improved the focus and fibrosis score in the salivary glands of the LAT mice. In vitro, stimulation with recombinant mouse CCL8 significantly increased the expression of collagen and ERK1/2 phosphorylation in NIH/3 T3. CONCLUSION: We clarified the overexpression and therapeutic potential of the mouse CCL8-CCR8 axis in LAT mice, which could play a crucial role in fibrosis via ERK1/2 phosphorylation, as well as the chemotaxis of inflammatory cells. The human CCL18-CCR8 axis might be a novel therapeutic target for IgG4-RD.

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.

P2Y2 receptor antagonism resolves sialadenitis and improves salivary flow in a Sjögren's syndrome mouse model.

OBJECTIVE: Sjögren's syndrome (SS) is a chronic autoimmune exocrinopathy characterized by lymphocytic infiltration of the salivary and lacrimal glands and decreased saliva and tear production. Previous studies indicate that the G protein-coupled P2Y2 nucleotide receptor (P2Y2R) is upregulated in numerous models of salivary gland inflammation (i.e., sialadenitis), where it has been implicated as a key mediator of chronic inflammation. Here, we evaluate both systemic and localized P2Y2R antagonism as a means to resolve sialadenitis in the NOD.H-2h4,IFNγ-/-,CD28-/- (NOD.H-2h4 DKO) mouse model of SS. DESIGN: Female 4.5 month old NOD.H-2h4 DKO mice received daily intraperitoneal injections for 10 days of the selective P2Y2R antagonist, AR-C118925, or vehicle-only control. Single-dose localized intraglandular antagonist delivery into the Wharton's duct was also evaluated. Carbachol-induced saliva was measured and then submandibular glands (SMGs) were isolated and either fixed and paraffin-embedded for H&E staining, homogenized for RNA isolation or dissociated for flow cytometry analysis. RESULTS: Intraperitoneal injection, but not localized intraglandular administration, of AR-C118925 significantly enhanced carbachol-induced salivation and reduced lymphocytic foci and immune cell markers in SMGs of 5 month old NOD.H-2h4 DKO mice, compared to vehicle-injected control mice. We found that B cells represent the primary immune cell population in inflamed SMGs of NOD.H-2h4 DKO mice that express elevated levels of P2Y2R compared to C57BL/6 control mice. We further demonstrate a role for P2Y2Rs in mediating B cell migration and the release of IgM. CONCLUSION: Our findings suggest that the P2Y2R represents a novel therapeutic target for the treatment of Sjögren's syndrome.

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.

Therapeutic effect of intraductal saline irrigation in chronic obstructive sialadenitis.

BACKGROUND: The aim of the present study was to evaluate the effectiveness of intraductal irrigation using normal saline in chronic obstructive sialadenitis. METHODS: Patients who had one of the following symptoms were recruited: pain, swelling, stiffness, and dry mouth. A total of 58 salivary glands in 33 patients were diagnosed as having sialadenitis using sialography and ultrasonography. The patients were divided into two groups (swelling group and dry mouth group), according to the major complaint. Repeated intraductal irrigation was performed on each gland. Difference of symptom severity evaluated using numerical rating scale (NRS), and ductal width measured using ultrasonography were compared between the two groups. RESULTS: The average NRS score was significantly decreased from 6.0 to 3.3 after 3-5 visits of intraductal irrigation (P < 0.05). The reduction in NRS was greater in the swelling group than in the dry mouth group, although the difference between the groups was not statistically significant. There was no change of ductal width before and after the irrigation. CONCLUSIONS: Intraductal irrigation according to this study method using normal saline is a simple treatment for the patients with chronic obstructive sialadenitis. It provides a conservative treatment option reducing the subjective symptoms.

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.

RORγt antagonist improves Sjögren's syndrome-like sialadenitis through downregulation of CD25.

OBJECTIVE: We reported previously that T-cell-specific RORγt-transgenic mice under human CD2 promoter (RORγt-Tg mice) developed severe spontaneous Sjögren's syndrome (SS)-like sialadenitis, induced by RORγt-overexpressing CD4+ T cells and reduced regulatory T cells. The purpose of this study was to clarify the effectiveness and mechanisms of action of A213, a RORγt antagonist, in RORγt-Tg mice with SS-like sialadenitis. METHODS: Six-week-old RORγt-Tg mice were administered orally of A213 or phosphate-buffered saline every 3 days for 2 weeks. We analyzed saliva volume, histopathology of salivary glands, populations of T cells in splenocytes and cervical lymph nodes (cLNs), and the protein expression levels of CD69 on CD4+ CD25+ Foxp3- and CD4+ CD25+ Foxp3+ cells in cLNs. We also investigated in vitro the potential immunomechanisms of action of A213. RESULTS: A213 significantly increased saliva volume, reduced mononuclear cell infiltration in salivary glands, and reduced the focus score of sialadenitis. Analysis of the immunomechanisms using cLNs showed A213 significantly reduced the proportion of CD4+ CD25+ /CD4+ T cells and the protein expression levels of CD69 on CD4+ CD25+ Foxp3- cells. In vitro experiments showed that A213 suppressed CD25 expression on CD4+ T cells and reduced IL-2 production from CD4+ T cells derived from RORγt-Tg mice. CONCLUSION: A213 improves SS-like sialadenitis through the inhibition of CD4+ CD25+ cells in cLNs.

Bilateral Neonatal Suppurative Sialadenitis Progressing to Abscess Formation in a Preterm Neonate

Aims We report a case of bilateral neonatal suppurative sialadenitis (NSS) in an extremely low birth weight infant (ELBW). Methods The infant developed bilateral sub-mandibular swelling at 3 weeks of age. NSS with abscess was confirmed with ultrasound. Despite intravenous antibiotic therapy the masses increased in size and developed abscesses. Results Unilaterally the abscess discharged via Wharton's duct necessitating intubation to protect the airway. The abscess remnant was incised and drained. Culture grew a methicillin sensitive staphyloccocus aureus. The NSS resolved following two weeks of antibiotics. Conclusion We wish to highlight the importance of early recognition of this rare condition in preterm neonates.