Rate of Submandibular Gland Atrophy Secondary to Chronic Sialolithiasis.

The rate of salivary gland atrophy secondary to chronic obstructive sialolithiasis has not been well-documented. The combination of 5 imaging studies over 12 years in a patient with repeat imaging for an unrelated pathology provides a unique opportunity to assess glandular atrophy over time. We hope that this case will support previous literature with an in vivo representation of the rate of glandular atrophy.

Sialography: a pictorial review.

Non-tumour inflammatory and obstructive salivary gland pathologies such as sialadenitis, sialolithiasis, sialadenosis, ductal strictures, etc. require precise radiological evaluation and mapping of salivary gland ductal system for better treatment outcome. Conventional sialography is considered as a useful and reliable technique in evaluation of salivary glands especially intrinsic and acquired abnormalities involving the ductal system and is useful for detection of non-radiopaque sialoliths which are invisible on routine plain radiographs. Primarily sialography is used as a diagnostic tool, additionally it plays an important therapeutic role as salivary gland lavage in cases of recurrent salivary gland infections and in obstructive salivary gland disorders by helping in clearance of mucous plugs or small sialoliths within the ducts. Recently, diagnostic performance of computed tomography (CT) sialography is being explored and has been reported to have high sensitivity in detection of small sialoliths and allows differentiation of sialoliths from other calcifications in glandular ductal system. Multiplanar three dimensional (3D) reconstructed CT images have been reported to play a key role in determination of anatomical location or extent of salivary gland disease without superimposition or distortion of structures. This review aims to discuss the disease specific applications of sialography and CT Sialography in particular for visualization of salivary gland disorders.

"Omepralith": A novel simulation model for training in sialoendoscopy.

INTRODUCTION: There are no previously described training models for learning or teaching how to remove lithiasis from the salivary ducts. Therefore, we present a new simulation model to enable us to faithfully represent the process of endoscopic lithiasis extraction by sialoendoscopy. MATERIALS AND METHODS: A simulation model was developed using a pig's head, omeprazole spheres were used to simulate lithiasis in the various ducts of each salivary gland and a Dormia basket was used to train in extraction of the lithiasis model. RESULTS: Twenty-seven residents in training and/or young specialists were successfully trained in this technique using this model. Twenty-six (96.3%) considered the model useful for training in the use of baskets; all of them were able to capture the omeprazole sphere in the salivary duct. A satisfaction rate of 92.25 out of 100 points was obtained through an anonymous survey. CONCLUSION: We describe a novel simulation model using omeprazole spheres, which allows the surgeon to practice how to diagnose and treat obstructive pathology of the salivary glands in a risk-free environment guaranteeing the reproducibility of the technique in conditions similar to those of normal practice.

IgG4-positive plasma cells in nonspecific sialadenitis and sialolithiasis.

Chronic sclerosing sialadenitis is commonly regarded as a manifestation of IgG4-related disease. We previously found that a high IgG4 expression or IgG4-related disease could accompany nonspecific sialadenitis, whereas chronic sclerosing sialadenitis was not directly associated with IgG4-related disease. Our previous findings lead us to hypothesize that these inflammatory conditions of the submandibular gland signify a continuous progression of disease rather than different disease entities. We, therefore, aimed to determine the presence of IgG4-positivity and genuine IgG4-related disease in a cohort of 165 submandibular gland specimens from patients who underwent surgery due to chronic nonspecific sialadenitis or sialolithiasis. To do so, we re-evaluated histopathological features and divided samples into three groups: (A) nonspecific sialadenitis without known sialolithiasis, (B) sialadenitis with sialolithiasis, and (C) sialolithiasis without sialadenitis. We performed immunohistochemical staining for IgG4, IgG, and CD31, and assessed the Boston consensus statement criteria for IgG4-related disease in IgG4-positive samples. We also reviewed patient records and supplemented follow-up data with a questionnaire among patients with IgG4-positive samples. IgG4-positive plasma cells (range 1-344) were found in 131 samples. Among these, 19 samples were classified as IgG4-positive (≥70 IgG4-positive plasma cells/high-power field). Two IgG4-positive samples were histologically highly suggestive of IgG4-related disease, but only one had a clinically confirmed diagnosis of IgG4-related disease. Our results indicate that patients with sialadenitis and sialolithiasis often present with IgG4-positive lymphoplasmacytic infiltrates, but exceedingly rarely present with genuine IgG4-related disease. In sialolithiasis without sialadenitis, IgG4-positive plasma cells are often absent or appear in small numbers. These results support our hypothesis of a continuum of disease, and indicate that progressive inflammation of the submandibular gland leads to the development of more specific pathological features over time.

«Omepralith»: un modelo de simulación novedoso para el entrenamiento en sialoendoscopia / “Omepralith”: A novel simulation model for training in sialoendoscopy

Introducción: No existen modelos de entrenamiento previamente descritos para aprender o enseñar cómo extraer litiasis de los conductos salivales. Es por ello que presentamos un novedoso modelo de simulación que nos permite representar fielmente el proceso de extracción endoscópica de litiasis mediante sialoendoscopia. Materiales y métodos: Se desarrolló un modelo de simulación utilizando la cabeza de un cerdo, se utilizaron esferas de omeprazol para simular la existencia de litiasis en los diversos conductos de cada glándula salival y se utilizó una cesta Dormia para entrenar la extracción del modelo de litiasis. Resultados: Veintisiete residentes en formación y/o jóvenes especialistas han entrenado con éxito esta técnica utilizando este modelo. Veintiséis (96,3%) consideraron útil el modelo para entrenar el uso de cestas, siendo todos ellos capaces de capturar la esfera de omeprazol en el conducto salival. Se obtuvo un porcentaje de satisfacción mediante una encuesta anónima de 92,25 sobre 100 puntos. Conclusión: Describimos un novedoso modelo de simulación mediante esferas de omeprazol, que permite al cirujano practicar cómo realizar el diagnóstico y tratamiento de la enfermedad obstructiva de glándulas salivales en un entorno libre de riesgos, garantizando la reproducibilidad de la técnica en condiciones similares a las de la práctica habitual.(AU)

Introduction: There are no previously described training models for learning or teaching how to remove lithiasis from the salivary ducts. Therefore, we present a new simulation model to enable us to faithfully represent the process of endoscopic lithiasis extraction by sialoendoscopy. Materials and methods: A simulation model was developed using a pig's head, omeprazole spheres were used to simulate lithiasis in the various ducts of each salivary gland and a Dormia basket was used to train in extraction of the lithiasis model. Results: Twenty-seven residents in training and/or young specialists were successfully trained in this technique using this model. Twenty-six (96.3%) considered the model useful for training in the use of baskets; all of them were able to capture the omeprazole sphere in the salivary duct. A satisfaction rate of 92.25 out of 100 points was obtained through an anonymous survey. Conclusion: We describe a novel simulation model using omeprazole spheres, which allows the surgeon to practice how to diagnose and treat obstructive pathology of the salivary glands in a risk-free environment guaranteeing the reproducibility of the technique in conditions similar to those of normal practice.(AU)

A Novel Approach for the Treatment of Sialolithiasis that Preserves Salivary Duct Anatomy.

OBJECTIVE: The sialendoscopy era in the treatment of salivary gland stones has reduced the use of classical surgical methods. However, the miniature ducts and tools may cause difficulties in removing large sialoliths. Therefore, invasive combined oral surgeries or gland resection may be considered. We searched for the most suitable method in order to stay in line with the minimally invasive approach that preserves the ductus anatomy, and that can reduce the surgical fears of patients. MATERIALS AND METHODS: The study included 84 cases (23 parotid and 61 submandibular) in whom stones were fragmented by pneumatic lithotripsy and removed between January 2015 and January 2020. The parotid cases comprised 7 females and 16 males, and the submandibular cases comprised 25 females and 36 males. Intraductal lithotripsy was performed using pneumatic lithotripter. This study has fourth level of evidence. RESULTS: Based on total number of cases (n = 84), success rate was 67/84 (79.7%) immediately after sialendoscopy, and overall success rate was 77/84 (91.6%). Based on number of stones treated (n = 111), our immediate success rate was 94/111 (84.6%), and overall success rate was 104/111 (93.7%). The success criteria were complete removal of the stone and fragments in a single sialendoscopy procedure and resolution of symptoms. CONCLUSIONS: We successfully treated salivary gland stones, including L3b stones, in our patient cohort with sialendoscopy combined with pneumatic lithotripsy. The lithotripsy method that we have adapted seems to be more useful and cost-effective compared to its alternatives. We were also able to preserve the ductus anatomy and relieve patients' concerns.Level of Evidence: Level IV.

Neutrophil Extracellular Traps Promote the Development and Growth of Human Salivary Stones.

Salivary gland stones, or sialoliths, are the most common cause of the obstruction of salivary glands. The mechanism behind the formation of sialoliths has been elusive. Symptomatic sialolithiasis has a prevalence of 0.45% in the general population, is characterized by recurrent painful periprandial swelling of the affected gland, and often results in sialadenitis with the need for surgical intervention. Here, we show by the use of immunohistochemistry, immunofluorescence, computed tomography (CT) scans and reconstructions, special dye techniques, bacterial genotyping, and enzyme activity analyses that neutrophil extracellular traps (NETs) initiate the formation and growth of sialoliths in humans. The deposition of neutrophil granulocyte extracellular DNA around small crystals results in the dense aggregation of the latter, and the subsequent mineralization creates alternating layers of dense mineral, which are predominantly calcium salt deposits and DNA. The further agglomeration and appositional growth of these structures promotes the development of macroscopic sialoliths that finally occlude the efferent ducts of the salivary glands, causing clinical symptoms and salivary gland dysfunction. These findings provide an entirely novel insight into the mechanism of sialolithogenesis, in which an immune system-mediated response essentially participates in the physicochemical process of concrement formation and growth.

A gland-sparing, intraoral sialolithotomy approach for hilar and intraparenchymal multiple stones in the submandibular gland.

Multiple intraglandular sialolithiasis for stones deep in the glandular parenchyma may require submandibulectomies, especially if sialendoscopic facilities are unavailable. We describe a gland-sparing intraoral sialolithotomy approach for both hilar and intraparenchymal multiple sialoliths. Nine patients with obstructive sialadenitis resulting from multiple sialoliths in both the deep hilar region and the submandibular gland parenchyma were selected for this study. Ultrasonography and computer tomography (CT) scans were performed to determine the location, number and sizes of the calculi and the distance between hilar and intraparenchymal sialoliths. All sialoliths were removed via gland-sparing, intraoral sialolithotomy. In all, 27 stones were found in the 9 patients. The hilar and deeper sialoliths were 4.5-11 and 0.8-4.5 mm, respectively, in diameter. The largest distance between the hilar and intraparenchymal sialoliths was 28.3 mm. Sialoliths in the hilar region were excised through an intraoral incision before deeper intraparenchymal stones were eased out of the same incision site. Postoperative follow-up imaging verified complete sialolith removal. Therefore, submandibular gland multiple sialoliths in the hilum and parenchyma can be successfully removed via an intraoral sialolithotomy under general anesthesia, thereby preserving the gland and restoring its secretory function.

Sialolithiasis of minor salivary gland in labial mucosa: a clinical finding / Sialolitíase de glândula salivar menor na mucosa labial: um achado clínico

Introduction: sialolithiasis is the most common form of obstructive sialadenitis caused by a mixture of different calcium phosphates and an organic matrix. It is one of the most common salivary gland diseases, often attributed to the submandibular gland, with no relation to age or gender. However, it is rarely reported in the minor salivary glands. Objective: the present study aims to report auncommon clinical finding case of a sialolithiasisof minor salivary gland in labial mucosa. Case report: a 43-year-old female patient presented with a single, yellow and asymptomatic nodule in the labial mucosa at clinical examination. The clinical hypotheses were lipoma and fibrous hyperplasia. The lesion was biopsied, and the histopathological analysis showed a mineralized tissue. The final diagnosis was sialolithiasis and the patient remained under follow-up (8 months) without relapse. Conclusion: this case shows that sialolithiasis should be included in the diagnostic hypotheses when occur in a minor salivary glands area and emphasizes the importance of a complete clinical examination since it was not complaint of the patient.(AU)

Introdução: a sialolitíase é a forma mais comum de sialadenite obstrutiva causada por um composto de diferentes produtos, como fosfato de cálcio e matriz orgânica. É uma das doenças mais comuns das glândulas salivares, geralmente atribuídas à glândula submandibular, sem relação com idade ou sexo. No entanto, raramente é relatada nas glândulas salivares menores. Objetivo: reportar um achado clínico incomum de sialolitíase em glândula oral menor na mucosa labial. Relato de caso: uma paciente do sexo feminino, de 43 anos, apresentou nódulo único, amarelo e assintomático na mucosa labial durante o exame clínico. As hipóteses clínicas foram lipoma e hiperplasia fibrosa. A lesão foi encaminhada para biópsia e a análise histopatológica mostrou um tecido mineralizado. O diagnóstico final foi de sialolitíase e o paciente permaneceu em acompanhamento por 8 meses sem recidiva. Conclusão: este caso mostra que a sialolitíase deve ser incluída nas hipóteses diagnósticas de lesões em áreas de glândulas salivares menores e enfatiza a importância de um exame clínico completo, pois não se tratava da queixa principal da paciente.(AU)

[Salivary gland cytopathology: Milan system 2018]. / Cytopathologie des glandes salivaires : le système de Milan 2018.

The salivary glands cytology is one of the most challenging area in cytopathology because of the wide diversity of benign and malignant tumors also because of their heterogeneity. However, fine needle aspiration cytology, with magnetic resonance imaging, represents a first-line examination to guide a possible surgical procedure and its extent. An accurate diagnosis of a specific tumor is sometimes difficult to assess in cytology. Also, as for gynecological, thyroid or urinary cytologies, a panel of experts met to develop a cytological classification of salivary gland lesions associated with a risk of malignancy and management proposals. The Milan System for Reporting Salivary Gland Cytopathology was published in 2018. The French Society of Clinical Cytology (SFCC) offers here an official summarized French version oh this terminology and recommends its use.

Intravascular Papillary Endothelial Hyperplasia With a Phlebolith of the Tongue: A Potential Pitfall.

Intravascular papillary endothelial hyperplasia (IPEH) or so-called Masson hemangioma is a benign nonneoplastic thrombus-associated organizing proliferation. De novo or secondary IPEHs are not uncommon oral lesions. An associated oral phlebolith is uncommon. Oral IPEHs and phleboliths can be diagnostically challenging. Certain histomorphologic features are helpful clues. Immunohistochemistry is a useful confirmatory tool. We present a case of an IPEH with phlebolith in a 37-year-old healthy lady who presented with a nodular lesion of the tongue. Histology showed a calcific nodule associated with an endothelial-lined vascular lesion with histomorphologic and immunohistochemical features of IPEH. The presence of a solitary extruded phlebolith could be challenging for pathologists and could be confused with other oral calculi, particularly sialoliths. The presence of papillary fronds, endothelial-lined minute vascular channels, fibrin thrombi, and hemosiderin pigment on the surface of the calculus points to a calcified thrombus. The lamellated concentric onion ring layers corresponding to Zahn lines around a central calcific nidus is another hint. Phleboliths should be considered by pathologists in their differential diagnosis of oral calculi. Distinction from sialolith is clinically important because the etiology and management are different.

Mineralization of Sialoliths Investigated by Ex Vivo and In Vivo X-ray Computed Tomography.

The fraction of organic matter present affects the fragmentation behavior of sialoliths; thus, pretherapeutic information on the degree of mineralization is relevant for a correct selection of lithotripsy procedures. This work proposes a methodology for in vivo characterization of salivary calculi in the pretherapeutic context. Sialoliths were characterized in detail by X-ray computed microtomography (µCT) in combination with atomic emission spectroscopy, Fourier transform infrared spectroscopy, X-ray diffraction, scanning electron microscopy, and transmission electron microscopy. Correlative analysis of the same specimens was performed by in vivo and ex vivo helical computed tomography (HCT) and ex vivo µCT. The mineral matter in the sialoliths consisted essentially of apatite (89 vol%) and whitlockite (11 vol%) with average density of 1.8 g/cm3. In hydrated conditions, the mineral mass prevailed with 53 ± 13 wt%, whereas the organic matter, with a density of 1.2 g/cm3, occupied 65 ± 10% of the sialoliths' volume. A quantitative relation between sialoliths mineral density and X-ray attenuation is proposed for both HCT and µCT.

Up-regulation of activation-induced cytidine deaminase and its strong expression in extra-germinal centres in IgG4-related disease.

Immunoglobulin (Ig) G4-related disease (IgG4-RD) is a systemic disorder involving benign mass formation due to fibrosis and intense lymphoplasmacytosis; the chronic inflammation associated with the disease might also contribute to oncogenesis. Activation-induced cytidine deaminase (AID), normally expressed in germinal centre activated B-cells, is an enzyme that edits DNA/RNA and induces somatic hypermutation and Ig class switching. AID expression is strictly controlled under physiological conditions; however, chronic inflammation and some infectious agents induce its up-regulation. AID is overexpressed in various cancers and may be important in chronic inflammation-associated oncogenesis. We examined AID expression in IgG4-related sialadenitis (n = 14), sialolithiasis (non-specific inflammation, n = 13), and normal submandibular glands (n = 13) using immunohistochemistry and quantitative real-time polymerase chain reaction (qPCR). Immunohistochemistry revealed significantly more AID-expressing cells in IgG4-related sialadenitis than in sialolithiasis or normal submandibular gland samples (P = 0.02 and P < 0.01, respectively); qPCR yielded similar results. Thus, AID was significantly more up-regulated and had higher expression in extra-germinal centres in IgG4-RD than in non-specific inflammation or normal conditions. This report suggests that IgG4-RD has several specific causes of AID up-regulation in addition to inflammation. Furthermore, chronic inflammation-associated AID-mediated oncogenesis is possible in IgG4-RD.

Adenoid Cystic Carcinoma With Sialolithiasis of the Left Submandibular Gland: A Case Report and Literature Review.

Adenoid cystic carcinoma is one of the most common salivary gland malignancies with poor long-term prognosis, but the coexistence of sialoliths is extraordinarily rare. In this article, we report a case of 30-year-old woman with a history of submandibular area swelling with intermittent pain increasing during mealtimes that had led her attending physician to diagnose a sialolith in the left submandibular gland on a radiograph 10 years before. However, the surgical specimen proved to be an adenoid cystic carcinoma accompanied with a sialolith. Histopathologically, the submandibular gland was displaced with a fibrous granulation tissue containing a small cribriform carcinoma invading the extracapsular region of the gland. We performed fluorescence in situ hybridization examination with an MYB-NFIB fusion probe of the lesion, with positive results. The patient underwent a supraomohyoid neck dissection as additional procedure because of the possibility of the extracapsular cancer nest remaining around the submandibular gland, but she remains well and disease free 11 years after the first operation.

One-Year symptom outcomes after sialolithiasis treatment with sialendoscopy-assisted salivary duct surgery.

OBJECTIVES: For chronic obstructive sialadenitis, there is a paucity of long-term prospective evidence of disease-specific symptom outcomes after sialendoscopy-assisted salivary duct surgery (SASDS). We prospectively assessed patients with sialolithiasis before and at 3 months and 1 year after SASDS using the Chronic Obstructive Sialadenitis Symptoms (COSS) questionnaire. METHODS: A prospective cohort study of adult patients with sialolithiasis who underwent SASDS and completed the COSS questionnaire over a 1-year follow-up period. The COSS questionnaire consists of 20 disease-specific symptom questions. COSS score outcomes were classified by complete, partial, and nonresolution, as well as by clinically significant improvement. RESULTS: Ninety-six patients underwent SASDS in 110 glands, of which 69 glands (63%) had findings of sialolithiasis. The following median COSS scores were reported for sialolithiasis glands: preoperative, 27.5 (interquartile range [IQR]: 12.5-44); 3 months, 1 (IQR: 0-5); and 1 year, 1 (IQR: 0-5.5). Patients with sialolithiasis reported statistically significant improvements in COSS scores from preoperative to 3 months post-SASDS (P > 0.05). A vast majority of patients maintained this improvement from 3 months to 1 year, indicating durability. Complete resolution of symptoms was attained in 95% and 85% of glands at 3 months and 1 year, respectively. Open/hybrid compared to endoscopic-only sialolithiasis extraction achieved similar 3-month and 1-year COSS outcomes. Potential risk factors for symptom persistence after SASDS included nonreachable parenchymal sialoliths and concurrent inflammatory disease and/or stenosis. CONCLUSION: SASDS for sialolithiasis extraction is associated with durable, long-term benefit regardless of endoscopic or open technique or sialolith location, suggesting that subsequent ductal stenosis from iatrogenic or sialolithiasis-related inflammation fibrosis is unlikely. LEVEL OF EVIDENCE: 4 Laryngoscope, 129:396-402, 2019.

Submandibular sialolithiasis: The roles of radiology in its diagnosis and treatment.

E. N was a 48-year-old man referred from a peripheral hospital to the Maxillofacial unit of Ahmadu Bello University Teaching Hospital, Zaria, on account of 1-year history of left-sided jaw pain and swelling, particularly after meal. The conventional plain radiograph of the jaws appeared normal as there was no opacity of soft tissue or calcific density visualized. Sialography revealed an oval-shaped filling defect in the dilated left Wharton's duct, which could suggest radiolucent calculus. Ultrasound scan showed the lesion as a brightly echogenic mass surrounded by anechoic clear fluid (saliva), casting posterior acoustic shadow. A diagnosis of left submandibular sialolithiasis was made. The maxillofacial surgeons decided to remove the calculus surgically, but the patient refused surgery and then defaulted from subsequent follow-up visits.

Résumé E. N était un homme âgé 48 ans visé d'un hôpital périphérique à l'unité maxillo faciale de l'Université Ahmadu Bello TeachingHospital, Zaria, en raison d'histoire 1 ans de douleur lamâchoire côté gauche et gonflement,particulier après repas. La plaine classique de radiographie mâchoires semblait normal car il n'y avait pas opacité des tissus mous ou densité calcifiée visualisée. Sialographie Révélé un défaut de remplissage forme ovale dans le conduit de la Wharton dilatée gauche,qui pourrait suggérer calcul radiotransparent. Échographie montré la lésion comme une masse échogène vive entourée par anéchoïque fluide clair (salive),moulage ombre acoustique postérieure. Un diagnostic a été fait de sialolithiases sous maxillaire gauche. Les chirurgiens maxillo décidé de supprimer le calcul chirurgicalement, mais le patient refuse chirurgie et visites suivi défaut de paiement en découlent. Mots-clés: Radiologie interventionnelle, sialographie, sialolithiases, ultrasons, conduit de Wharton.

Lack of evidence that nephrolithiasis increases the risk of sialolithiasis: A longitudinal follow-up study using a national sample cohort.

OBJECTIVE: The objective of this study was to evaluate the risk of sialolithiasis in nephrolithiasis patients. METHODS: Using data from the national cohort study from the Korean Health Insurance Review and Assessment Service, we selected 24,038 patients with nephrolithiasis. The control group consisted of 96,152 participants without nephrolithiasis who were matched 1:4 by age, sex, income, region of residence, diabetes, hypertension, and dyslipidemia. The incidence of sialolithiasis in the two groups was compared, with a follow-up period of up to 12 years. The crude and adjusted hazard ratio (HR) of nephrolithiasis to sialolithiasis was analyzed with a Cox-proportional hazard regression model. RESULTS: The rates of sialolithiasis in the nephrolithiasis group and the control group were not significantly different (0.08% vs. 0.1%, P = 0.447). The crude and adjusted hazard ratios of nephrolithiasis to sialolithiasis were not statistically significant (crude HR = 0.82, 95% confidence interval [CI] = 0.50-1.35, P = 0.448; adjusted HR = 0.81, 95% CI = 0.49-1.33, P = 0.399). Subgroup analyses according to age and sex also failed to reveal statistical significance. CONCLUSION: There is no evidence of an increased risk of sialolithiasis associated with nephrolithiasis. We suggest that routine evaluation for sialolithiasis in all patients with nephrolithiasis is not necessary.