Search Results (12)

Objective: Recent evidence has suggested that sialendoscopy should be the first-line treatment for chronic sialadenitis. This study aimed to assess the efficacy of steroid irrigation during sialendoscopy in non-lithiasic chronic sialadenitis. Methods: We conducted a systematic search of the databases of PubMed, Scopus, and Web of Science up to the 3rd of November 2024 for completed studies investigating the efficacy of steroid irrigation during sialendoscopy for chronic non-lithiasic sialadenitis. Primary outcomes were the number of patients with recurrence of sialadenitis symptoms and the number of patients who required a revision sialendoscopy. Secondary outcomes included the assessment of major complications of the procedure. Random-effect meta-analysis of proportion was conducted using Open Meta-Analyst software. Additionally, separate subgroup analyses linked to the underlying salivary gland disease were carried out. The quality of the included studies was evaluated utilizing the Moga tool and the ROBINS-I tool. Results: Thirty studies qualified for inclusion in the present systematic review. The weighted pooled proportion of recurrence after sialendoscopy by patient was 27.49% (95% CI: 21.04–34.45), whereas the revision rate was 10.64% (95% CI: 7.74–13.93). In every subgroup, the proposed intervention exhibited adequate efficacy for the symptomatic relief of patients, with similar rates of revision and repetition. The rate of major complications per patient was 0.77%. Conclusions: This study demonstrates that interventional sialendoscopy with intraductal steroid irrigation may yield therapeutic benefit for patients with chronic non-lithiasic sialadenitis and could be considered a safe treatment. Full article

Background: Kuttner’s tumour refers to chronic sclerosing sialadenitis commonly affecting the submandibular gland. It clinically mimics submandibular gland neoplasms and has a possible association with IgG4-related disease. The literature on standardised diagnostic and management pathways is limited. This paper presents a case series and narrative literature review to support a proposed diagnostic and management approach for Kuttner’s tumour. Methods: Twelve cases of patients with a Kuttner’s tumour aged between 55 and 87, identified from 2018 to 2025 through hospital records, were reviewed. All patients underwent ultrasound assessment using standardised diagnostic criteria and were followed up clinically and radiologically every six months. In parallel, we performed a narrative review of studies published between 2005 and 2025, identifying nine relevant articles to contextualise our findings. Results: Our 12-patient case series highlights the potential for Kuttner’s tumour to progress to bilateral involvement and IgG4-related disease. Most cases resolved spontaneously with ultrasound-led monitoring. One progressed to IgG4-RD and responded to glucocorticoids. Our findings support the selective use of invasive tests, baseline serum IgG4 testing, and a six-monthly follow-up strategy. Despite similarities within the existing literature, international variation highlights the need for standardised diagnostic and management protocols. Conclusions: We recommend a conservative, structured approach to managing Kuttner’s tumour, with six-monthly clinical and radiological follow-ups. Based on one case progressing to multi-organ IgG4-related disease, we now recommend routinely measuring serum IgG4 concentrations at diagnosis. The role of magnetic resonance imaging, fine-needle aspiration cytology, and biopsy should be considered on a case-by-case basis. Further research is needed to validate this approach and assess long-term outcomes. Full article

Background/Objectives: Primary Sjogren’s syndrome (pSS) is an autoimmune disease caused by chronic inflammation of the exocrine glands. We aimed to determine the immunohistochemical parameters that may help in the diagnosis of pSS and to determine histopathological markers for the early diagnosis of patients. Methods: Hematoxylin–eosin-stained preparations of salivary gland tissue samples of a control group consisting of 9 healthy patients and 12 patients diagnosed with non-specific chronic sialadenitis (NSCS) and a study group consisting of 39 patients diagnosed with pSS were evaluated. SSA/Ro (TRIM21), SSB/la, BAFF, enolase (ENO1), and MMP-9 antibodies were applied to the samples to assess the presence of staining in the ductal, acini, and inflammation regions. Results: In our study, mild staining with the SSA/Ro (TRIM21) antibody was observed in the ductal areas of all pSS cases, while no staining was detected in the healthy and NSCS cases (p < 0.01). Additionally, stronger staining was identified in the ductal and inflammatory areas of the pSS cases with BAFF compared to the control group. The staining for the ENO1 antibody was higher and more intense in the NSCS and pSS cases compared to those with normal salivary glands, and this difference was found to be statistically significant (p < 0.01). While mild staining was detected in the ductal areas with MMP-9 antibody in most of the NSCS and pSS cases, it was detected in 11% of the healthy cases. Conclusions: Our study suggests that SSA/Ro (TRIM21), ENO1, MMP9, and BAFF can be used to confirm the diagnosis in cases of suspicion. Full article

Post-irradiation xerostomia remains a significant quality of life concern for patients with head and neck cancers. Conventional therapies offer limited effectiveness. This study aims to investigate the therapeutic potential of office-based salivary ductal steroid irrigation in patients with post-irradiation xerostomia. This single-center observational study recruited 147 head and neck cancer patients suffering from post-irradiation xerostomia between November 2020 and October 2022. All included subjects received at least one round of successful salivary ductal cannulation and irrigation. The primary measure of efficacy was improvement in subjective xerostomia and objective salivary amylase levels. A logistic regression was employed to evaluate factors affecting treatment responsiveness. The response rate among nasopharyngeal cancer (NPC) patients was 74.8%, and that among non-NPC cancer was 65.6%, without significant intergroup differences. The statistical analysis revealed no significant influence of age, gender, or disease stage on treatment responsiveness. Post-treatment salivary amylase levels were significantly higher in responsive non-NPC patients. In conclusion, salivary ductal steroid irrigation emerged as a promising therapeutic modality for the management of post-irradiation xerostomia in head and neck cancer patients. While no explicit factors were predictive of responsiveness, the high rate of symptom improvement suggests that this therapy may be a viable alternative for patients that are refractory to standard treatments. Full article

Saliva is an irritant of the subcutaneous tissue, thus causing the development of a non-epithelial reactive pseudocapsule. Metaplastic ossification of the pseudocapsule is a condition rarely described in the veterinary literature. The main causes of calcification are trauma, tumours, various chronic inflammatory conditions and fibrodysplasia ossificans progressiva. The aim of the present case series was to describe three dogs affected by a calcified salivary mucocele. The medical records of dogs affected by a cervical sialocele were retrospectively evaluated, and three cases met the inclusion criteria. All the dogs in this study were referred to the Veterinary Teaching Hospital (VTH) of the Department of Veterinary Sciences of the University of Turin (Turin, Italy) for a large solid mass in the intermandibular region. The diagnosis of a mucocele was confirmed clinically by centesis and by radiography or CT. Complete excision of both the pseudocyst and the ipsilateral mandibular/monostomatic sublingual salivary gland was performed in all cases. The histological report showed large areas of bone metaplasia within the pseudocapsule and chronic sialadenitis. Based on this limited case series, complete excision of the pseudocyst and a concurrent sialoadenectomy provided an effective treatment for this rare salivary mucocele disorder. Full article

This review highlights oral anomalies with major clinical impact in Addison disease (AD), including dental health and dermatologic features, through a dual perspective: pigmentation issues and AD comorbidities with oral manifestations. Affecting 92% of AD patients, cutaneomucosal hyperpigmentation is synchronous with or precedes general manifestations by up to a decade, underlying melanocytic infiltration of the basal epidermal layer; melanophages in the superficial dermis; and, rarely, acanthosis, perivascular lymphocytic infiltrate, and hyperkeratosis. Intraoral pigmentation might be the only sign of AD; thus, early recognition is mandatory, and biopsy is helpful in selected cases. The buccal area is the most affected location; other sites are palatine arches, lips, gums, and tongue. Pigmented oral lesions are patchy or diffuse; mostly asymptomatic; and occasionally accompanied by pain, itchiness, and burn-like lesions. Pigmented lingual patches are isolated or multiple, located on dorsal and lateral areas; fungiform pigmented papillae are also reported in AD individuals. Dermoscopy examination is particularly indicated for fungal etiology; yet, it is not routinely performed. AD’s comorbidity burden includes the cluster of autoimmune polyglandular syndrome (APS) type 1 underlying AIRE gene malfunction. Chronic cutaneomucosal candidiasis (CMC), including oral CMC, represents the first sign of APS1 in 70–80% of cases, displaying autoantibodies against interleukin (IL)-17A, IL-17F ± IL-22, and probably a high mucosal concentration of interferon (IFN)-γ. CMC is prone to systemic candidiasis, representing a procarcinogenic status due to Th17 cell anomalies. In APS1, the first cause of mortality is infections (24%), followed by oral and esophageal cancers (15%). Autoimmune hypoparathyroidism (HyP) is the earliest endocrine element in APS1; a combination of CMC by the age of 5 years and dental enamel hypoplasia (the most frequent dental complication of pediatric HyP) by the age of 15 is an indication for HyP assessment. Children with HyP might experience short dental roots, enamel opacities, hypodontia, and eruption dysfunctions. Copresence of APS-related type 1 diabetes mellitus (DM) enhances the risk of CMC, as well as periodontal disease (PD). Anemia-related mucosal pallor is related to DM, hypothyroidism, hypogonadism, corresponding gastroenterological diseases (Crohn’s disease also presents oral ulceration (OU), mucogingivitis, and a 2–3 times higher risk of PD; Biermer anemia might cause hyperpigmentation by itself), and rheumatologic diseases (lupus induces OU, honeycomb plaques, keratotic plaques, angular cheilitis, buccal petechial lesions, and PD). In more than half of the patients, associated vitiligo involves depigmentation of oral mucosa at different levels (palatal, gingival, alveolar, buccal mucosa, and lips). Celiac disease may manifest xerostomia, dry lips, OU, sialadenitis, recurrent aphthous stomatitis and dental enamel defects in children, a higher prevalence of caries and dentin sensitivity, and gingival bleeding. Oral pigmented lesions might provide a useful index of suspicion for AD in apparently healthy individuals, and thus an adrenocorticotropic hormone (ACTH) stimulation is useful. The spectrum of autoimmune AD comorbidities massively complicates the overall picture of oral manifestations. Full article

Sjögren’s syndrome (SS) is a chronic autoimmune disease characterized by lymphocytic infiltrates in the salivary and lachrymal glands resulting in oral and ocular dryness. There are no clinically approved therapies to slow the progression of SS. Immune cells possess receptors for the neurotransmitter GABA (GABA-Rs) and their activation has immunoregulatory actions. We tested whether GABA administration has potential for amelioration of SS in NOD.B10-H2^b and C57BL/6.NOD-Aec1Aec2 mice, two spontaneous SS models. Oral GABA treatment was initiated (1) after the development of sialadenitis but before the onset of overt symptoms, or (2) after the appearance of overt symptoms. When assessed weeks later, GABA-treated mice had greater saliva and tear production, as well as quicker times to salvia flow, in both SS mouse models. This was especially evident when GABA treatment was initiated after the onset of overt disease. This preservation of exocrine function was not accompanied by significant changes in the number or area of lymphocytic foci in the salivary or lachrymal glands of GABA-treated mice and we discuss the possible reasons for these observations. Given that GABA-treatment preserved saliva and tear production which are the most salient symptoms of SS and is safe for consumption, it may provide a new approach to help ameliorate SS. Full article

Sialolithiasis is a chronic disease in which a sialolith (salivary stone) causes recurrent inflammation of the affected salivary gland. Anemia of inflammation is a well-described pathology in which a chronic inflammatory disease leads to a reduction in the red blood cell count, hemoglobin and hematocrit values. In this retrospective cohort study, we aim to find whether removal of the sialolith and alleviation of the inflammation affect the complete blood count results. We examined data regarding forty-nine patients who underwent surgery for the removal of a submandibular gland sialolith using the duct-stretching technique. Complete blood counts two years before and after the surgical procedure were collected. The average pre-procedure and post-procedure values were calculated for each patient to establish the average blood profile. The pre- and post-procedure values were compared to evaluate the effect of the surgical treatment on the blood profile. We found that the average blood count values for patients with sialolithiasis were towards the lower end of the normal range. Post-surgery, a significant increase in hematocrit, hemoglobin and red blood cell count was observed, which was more pronounced in the older age group and in patients with co-morbidities. We conclude that sialolith removal surgery is associated with significant improvement in the complete blood count values, especially in the elderly and in patients and with co-morbidities. The speculated pathogenesis is relative anemia of inflammation. Full article

Sjögren’s Syndrome (SS), a chronic autoimmune disorder affecting multiple organ systems, is characterized by an elevated type I interferon (IFN) response. Activation of Stimulator of Interferon Genes (STING) protein induces type I IFN and in mice, several features of SS, including anti-nuclear antibodies, sialadenitis, and salivary gland dysfunction. Since lung involvement occurs in one-fifth of SS patients, we investigated whether systemic activation of STING also leads to lung inflammation. Lungs from female C57BL/6 mice injected with the STING agonist 5, 6-Dimethylxanthenone-4-acetic acid (DMXAA), were evaluated for acute and chronic inflammatory responses. Within 4h of DMXAA injection, the expression of Ifnb1, Il6, Tnf, Ifng, and Mx1 was significantly upregulated. At 1 and 2 months post-treatment, lungs showed lymphocytic infiltration in the peri-bronchial regions. The lungs from DMXAA treated mice showed an increased expression of multiple chemokines and an increase in lymphatic endothelial cells. Despite STING expression in bronchial epithelium and cells lining the alveolar wall, bone marrow chimeras between STING knockout and wild type mice showed that STING expression in hematopoietic cells was critical for lung inflammation. Our results suggest that activation of the STING pathway might be involved in SS patients with concomitant salivary gland and lung disease. Full article

A preliminary evaluation of the parotid secretion cellular composition in patients with Sjogren’s Syndrome (SS) and a diagnostic accuracy assessment of salivary lymphocyte detection and immunophenotyping in Sjogren’s Syndrome diagnosis and prognosis were performed. The study included 40 consecutive patients, aged 19–60 years, with parenchymal sialadenitis associated with Sjogren’s Syndrome, and 20 healthy donors. The exclusion criteria were exacerbation of sialadenitis, chronic infections, malignant neoplasms, and lymphoproliferative diseases. The standard diagnostic tests were minor salivary gland biopsy and parotid sialography. Immunophenotyping of parotid secretion lymphocytes was performed by multicolor flow cytometry. Lymphocytes were detectable in parotid secretion of patients affected by Sjogren’s Syndrome, both primary (pSS) and secondary (sSS) form, but not in that from healthy donors. Sensitivity, specificity, positive, and negative predictive values of lymphocytes detection in parotid saliva were 77.5%, 100%, 100%, and 69%, respectively. The mean numbers of the total T-cell population, T-helper cells, and T-cytotoxic cells were 71.7%, 41.6%, and 53%, respectively. The immunophenotype of lymphocytes obtained by patients’ parotid flow resembles the immunophenotypes of glandular biopsies currently known. Our preliminary data suggest the use of saliva as an alternative and non-invasive method for evaluating the prognosis of Sjogren’s Syndrome. Full article

Autoimmune pancreatitis (AIP), a unique subtype of pancreatitis, is often accompanied by systemic inflammatory disorders. AIP is classified into two distinct subtypes on the basis of the histological subtype: immunoglobulin G4 (IgG4)-related lymphoplasmacytic sclerosing pancreatitis (type 1) and idiopathic duct-centric pancreatitis (type 2). Type 1 AIP is often accompanied by systemic lesions, biliary strictures, hepatic inflammatory pseudotumors, interstitial pneumonia and nephritis, dacryoadenitis, and sialadenitis. Type 2 AIP is associated with inflammatory bowel diseases in approximately 30% of cases. Standard therapy for AIP is oral corticosteroid administration. Steroid treatment is generally indicated for symptomatic cases and is exceptionally applied for cases with diagnostic difficulty (diagnostic steroid trial) after a negative workup for malignancy. More than 90% of patients respond to steroid treatment within 1 month, and most within 2 weeks. The steroid response can be confirmed on clinical images (computed tomography, ultrasonography, endoscopic ultrasonography, magnetic resonance imaging, and ¹⁸F-fluorodeoxyglucose-positron emission tomography). Hence, the steroid response is included as an optional diagnostic item of AIP. Steroid treatment results in normalization of serological markers, including IgG4. Short- and long-term corticosteroid treatment may induce adverse events, including chronic glycometabolism, obesity, an immunocompromised status against infection, cataracts, glaucoma, osteoporosis, and myopathy. AIP is common in old age and is often associated with diabetes mellitus (33–78%). Thus, there is an argument for corticosteroid therapy in diabetes patients with no symptoms. With low-dose steroid treatment or treatment withdrawal, there is a high incidence of AIP recurrence (24–52%). Therefore, there is a need for long-term steroid maintenance therapy and/or steroid-sparing agents (immunomodulators and rituximab). Corticosteroids play a critical role in the diagnosis and treatment of AIP. Full article

Ultrasonography has become a valuable tool for the assessment of salivary gland involvement in Sjögren syndrome. Diagnostic pitfalls can be encountered in late stages of the disease, in which morphologic changes may overlap with other pathologies of the salivary glands. We present the case of a female Sjögren syndrome patient with lack of significant sicca symptoms and unilateral occurrence of parotidomegaly, which prompted the suspicion of a parotid gland tumor. Due to the atypical clinical profile, diagnosis of Sjögren syndrome was delayed, at which point, severe sialadenitis produced a cystic transformation of the parotid gland parenchyma. Full article