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.

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.

Development and accuracy validation of a fat fraction imaging biomarker for sialadenitis in the parotid gland.

BACKGROUND: The diagnosis of sialadenitis, the most frequent disease of the salivary glands, is challenging when the symptoms are mild. In such cases, biomarkers can be used as definitive diagnostic indicators. Recently, biomarkers have been developed by extracting and analyzing pathological and morphological features from medical imaging. This study aimed to establish a diagnostic reference for sialadenitis based on the quantitative magnetic resonance imaging (MRI) biomarker IDEAL-IQ and assess its accuracy. METHODS: Patients with sialadenitis (n = 46) and control subjects (n = 90) that underwent MRI were selected. Considering that the IDEAL-IQ value is a sensitive fat fractional marker to the body mass index (BMI), all subjects were also categorized as under-, normal-, and overweight. The fat fraction of parotid gland in the control and sialadenitis groups were obtained using IDEAL-IQ map. The values from the subjects in the control and sialadenitis groups were compared in each BMI category. For comparison, t-tests and receiver operating characteristic (ROC) curve analyses were performed. RESULTS: The IDEAL-IQ fat faction of the control and sialadenitis glands were 38.57% and 23.69%, respectively, and the differences were significant. The values were significantly lower in the sialadenitis group (P), regardless of the BMI types. The area under the ROC curve (AUC) was 0.83 (cut-off value: 28.72) in patients with sialadenitis. The AUC for under-, normal-, and overweight individuals were 0.78, 0.81, and 0.92, respectively. CONCLUSIONS: The fat fraction marker based on the IDEAL-IQ method was useful as an objective indicator for diagnosing sialadenitis. This marker would aid less-experienced clinicians in diagnosing sialadenitis.

Value of High-Frequency Ultrasonography in the Qualitative and Semi-Quantitative Assessment of Immunoglobulin G4-Related Submandibular Sialadenitis.

OBJECTIVE: To assess the value of high-frequency ultrasonography in the evaluation of immunoglobulin G4-related submandibular sialadenitis (IgG4-SS). METHODS: Thirty-four submandibular glands in 17 patients with IgG4-SS were retrospectively enrolled, as well as 34 submandibular glands in 17 healthy control subjects. Qualitative ultrasonic features including submandibular gland size, border, echogenicity, and vascularity were reviewed. Two different scoring systems (0-16 and 0-48, respectively) were used for semi-quantitative analysis of imaging features. Comparison of both qualitative and semi-quantitative ultrasonic analysis were made between patients with IgG4-SS and healthy controls. Spearman correlation was used to explore relationships between variables. RESULTS: The submandibular glands with IgG4-SS presented with enlarged size, rough border, increased vascularity, and abnormal echogenicity (All P < .05). The most common echogenicity pattern for IgG-SS was diffuse hypoechoic foci pattern (44.1%), followed by superficial hypoechoic pattern (20.6%), tumor-like pattern (14.7%), and diffuse hypoechogenicity pattern (11.8%). Most IgG4-SS glands presented linear hyperechogenicity in parenchyma (91.2%). Based on both scoring system, scores of IgG4-SS were significantly higher than those of the controls (All P < .05). Association analysis of both scoring systems showed positive correlation of scores with vascularity in the gland parenchyma (All P < .05). CONCLUSION: The ultrasonic features of IgG4-SS comprise enlarged gland, rough border, increased vascularity, and abnormal echogenicity, which correlate with its pathological characteristics. The most common echogenicity pattern for IgG4-SS was diffuse hypoechoic foci pattern. Semi-quantitative analysis systems could be useful in the assessment of IgG4-SS. Ultrasound is a potential, valuable, and non-invasive tool for the diagnosis and evaluation of IgG4-SS.

Management of iodine contrast induced salivary gland swelling (sialadenitis): experiences from an observational study.

BACKGROUND: Iodine contrast-induced sialadenitis (CIS) is an uncommon adverse effect to iodine-containing contrast exposition. There is scarce literature about its clinical course and demography. OBJECTIVES: (1) To determine the clinical course and management of CIS. (2) To understand if CIS might be as rare as reported. MATERIAL AND METHODS: A 2-month prospective observational study was conducted in the emergency room of a tertiary institution. ENT physicians on call received clinical formation about CIS while emergency physicians did not. During the study period, patients admitted at the emergency room matching the clinical and radiological features of CIS, were included. Information about demographics, iodine exposure, diagnostic workup, clinical course, and management was analyzed. RESULTS: ENT physicians on call detected 4 cases, however, emergency physicians did not. Patients were aged 68-76 years and presented a bilateral submandibular gland swelling debuting 12 to 72 h after an exposure to iodinated contrast. Characteristic ultrasonographic findings supported the diagnosis and the clinical course was self-limited after 60 to 150 h. CONCLUSIONS AND SIGNIFICANCE: Physicians' familiarity with CIS allows its detection and avoids costly and potentially harmful therapeutic/diagnostic efforts. The incidence is probably underestimated; however, further incidence studies are needed.

Ultrasound scoring systems affect the distribution of sialadenitis scores in Sjögren's syndrome: an inter-system reproducibility study.

OBJECTIVES: Salivary gland ultrasonography (SGUS) is commonly employed in the diagnosis and follow-up of patients with Sjögren's syndrome (SS) and multiple scoring systems have been developed to quantify the grade of sialadenitis of major salivary glands (SG). Their diagnostic performance seems overall comparable, however, the parameters evaluated by the various systems are different. The objective of this study was to compare how four different scoring systems affect the distribution of sialadenitis grades. METHODS: One hundred and three SGUS images from 26 SS patients were blindly scored by two investigators according to the De Vita, Salaffi, Milic and OMERACT scoring systems in independent sessions. RESULTS: The distribution of SGUS images according to De Vita, Salaffi, Milic and OMERACT systems was significantly different. At post-hoc analysis, Milic system performed differently compared to the De Vita (p<0.0001), OMERACT (p<0.0001) and Salaffi (p<0.0001) systems, showing a relative overestimation of sialadenitis grade. CONCLUSIONS: Milic scoring system showed to relatively overestimate the grade of sialadenitis compared to De Vita, Salaffi and OMERACT systems. Although all scoring systems seem to be comparable in terms of diagnostic accuracy, in the prospect of selecting one system to be potentially included in future versions of SS classification criteria, it is important to compare their ability to classify SGUS images among the various degrees of sialadenitis.

Epidemiologic, radiologic, and sialendoscopic aspects in chronic obstructive sialadenitis.

PURPOSE: Chronic obstructive sialadenitis (COS) is a recurring inflammation of the salivary gland. To date, there are no known predisposing factors for COS. Given the advances seen in radiology and sialendoscopy, we must update our knowledge of COS, analyzing factors that can favor its development. METHODS: We prospectively analyzed 333 patients who underwent sialendoscopy between 2012 and 2021. Epidemiologic, radiologic, and sialendoscopy-related factors were correlated. Suspected diagnosis was established based on the clinical and radiologic data. The final diagnosis was determined on the basis of sialendoscopic findings. RESULTS: The most common etiology of COS was stricture (40.8%). Lack of papilla distensibility (LPD) was also described as an etiology. COS was related to patient gender and age. Submandibular gland involvement was significantly more associated with lithiasis and LPD, while COS of the parotid gland was most frequently caused by stricture. Radioiodine sialadenitis and Sjögren's syndrome were significantly associated with stricture. MR sialography (MR-Si) showed the best overall sensitivity and specificity. CONCLUSION: In our series, stricture was the most common cause of COS. We describe LPD as a frequent cause of COS in this series; ours is the first study to report this finding. There was a significant association between the salivary gland involved, patient sex and age, and the cause of COS. MR-Si showed the greatest diagnostic yield.

Chronic sclerosing sialadenitis (Küttner's tumour) of the submandibular gland: a neoplastic mimic.

A 74-year-old man was referred for a plastic surgery consultation for two previous acute episodes of right submandibular sialadenitis. Physical examination revealed a lump in the right submandibular region, painful on palpation. The initial impression on clinical examination was of sialolithiasis. The sonography demonstrated a structural heterogeneity of the submandibular gland with a hypoechogenic and vascularised nodular formation (1.5×1.2 cm), suggestive of a mixed tumour of the right maxillary gland, requiring histological evaluation. He underwent a right submandibulectomy and an IgG4-positive chronic sclerosing sialadenitis was diagnosed. The patient's condition evolved favourably, resulting in a full recovery. The clinical features and differential diagnosis of this entity are debated in light of relevant literature.

The applicability of ultrasound in the diagnosis of inflammatory and obstructive diseases of the major salivary glands: a scoping review.

OBJECTIVES: The purpose of the present scoping review was to determine the contribution of ultrasound images in the diagnosis of inflammatory and obstructive diseases of the major salivary glands (MSGs). METHODS: A search of studies of ultrasonographic assessments of human samples was performed in several electronic databases and grey literature up to July 2021. The extracted data were the examined MSG; the diagnostic value of ultrasound (sensibility, specificity, positive- and negative predictive value, accuracy); features of lesions, including number, echogenicity, echotexture, form, margins, size, posterior acoustic aspect, and location; and related clinical information, such as swelling, palpation, sensible to pain, salivation, lymph nodes, recurrence, duration, and causes. RESULTS: After verifying the eligibility criteria, 90 articles focused on detecting inflammatory, and obstructive diseases of the MSG were gathered, with variable study designs and size samples. A wide variety of pathologies were assessed, including sialolitiasis (n = 45), acute sialadenitis (n = 30), chronic sialadenitis (n = 25), granulamatous diseases (n = 15), Kuttner's tumor (n = 11), juvenile recurrent parotitis (n = 9), abscess (n = 7), post-radiotherapy sialadenitis (n = 6), sialadenosis (n = 9), abscess (n = 7), IgG4-related disease sialadenitis (n = 5), HIV-sialadenitis (n = 4), obstructive sialadenitis (n = 3), iodinated contrast-induced sialadenitis (n = 2), and pneumoparotitis (n = 1). Most studies were case reports or series of cases. Few studies exhibited data about the accuracy of ultrasound in detecting MSG diseases. CONCLUSIONS: The present scoping review concluded that ultrasound aspects of different MSG pathologies are similar but contribute to their differential diagnosis and can be considered as a valuable initial method for assessing the MSG of adults and children.

A case of acute bilateral iodine-induced submandibular sialadenitis: Ultrasound findings.

Acute sialadenitis is a rare adverse reaction to iodine-based contrast agents. Ultrasound (US) is usually the preferred imaging method to evaluate the salivary glands; along with clinical and anamnestic data, US allows the diagnosis of contrast-induced sialadenitis. We present a case of acute bilateral submandibular sialadenitis induced by intravenous administration of iodine-based contrast media for a contrast-enhanced computed tomography scan diagnosed by US.

Sonographic findings of immunoglobulin G4-related sialadenitis and differences from Sjögren's syndrome.

OBJECTIVE: To evaluate ultrasonic features of the major salivary glands in patients with immunoglobulin G4-related sialadenitis (IgG4-RS) and to explore the differences between IgG4-RS and Sjögren's syndrome (SS). METHOD: We conducted the study in 150 patients with IgG4-RS and 100 patients with SS. Ultrasonographic variables of the static images of major salivary glands were analysed. An experienced radiologist scored the confidence rating regarding the presence of the characteristic imaging findings using a five-grade rating system. Ultrasonography scores between IgG4-RS and SS were compared. RESULTS: The major salivary glands were significantly larger in patients with IgG4-RS than in the SS group. The main features of ultrasonography of the salivary glands in IgG4-RS were various hypoechoic lesions and increased colour Doppler signalling. In contrast, the major salivary glands in SS exhibited hyperechoic lines and/or spots and obscuration of the gland configuration. The scores of the summarized sonographic characteristics also showed statistically significant differences between the IgG4-RS and SS groups. CONCLUSION: This study revealed different ultrasonic features of the major salivary glands in patients with IgG4-RS and SS. The scored sonographic features were helpful in differentiating IgG4-RS from SS. Consequently, we suggest that ultrasonography of major salivary glands could be a useful imaging procedure in the evaluation of patients suspected of having IgG4-RS.

Reticular enhancement of the submandibular gland on contrast-enhanced magnetic resonance imaging in three cases with IgG4-related chronic sclerosing sialadenitis.

The present case study reports contrast-enhanced magnetic resonance findings in three patients with histopathologically proven IgG4-related chronic sclerosing sialadenitis of the submandibular gland. All three patients presented with painless swelling of the submandibular region. The contrast-enhanced T1-weighted images showed reticular enhancement of the swollen submandibular gland. Radiological-pathological correlation revealed that the characteristic reticular enhancement corresponded to fibrosis and to inflammatory cell infiltration in the interlobular septa and in the periductal region of the submandibular gland.

Ultrasonography of IgG4-related dacryoadenitis and sialadenitis: Imaging features and clinical usefulness.

OBJECTIVES: To clarify the ultrasonographic features of immunoglobulin G4 (IgG4)-related dacryoadenitis and sialadenitis (IgG4-DS) and their usefulness in clinical diagnostic sessions. METHODS: By re-evaluating 96 consecutive patients with IgG4-related disease, we identified 54 patients (male:female = 37:17; median age, 69.5 years) who underwent lacrimal or submandibular gland (LG or SG, respectively) ultrasonography and computed tomography (CT). Their clinical and ultrasonographic features were retrospectively analysed. Radio-pathological correlations were also examined in LG (23 cases) and SG lesions (20 cases). Additionally, the diagnostic accuracy of CT for LG/SG lesions was evaluated. RESULTS: Abnormal ultrasonographic findings were detected in 33 (LGs) and 38 (SGs) patients, and most of them were observed bilaterally. All lesions were well demarcated and demonstrated diffuse low-echoic areas (rocky pattern) or multiple low-echoic nodules surrounded by high-echoic linear shadows (cobblestone pattern) corresponding to intra-lobular inflammation and inter-lobular fibrosis. Moreover, 42% (LGs; 14/33) and 42% (SGs; 16/38) patients had glandular lesions without clinical symptoms associated with the affected glands. The diagnostic accuracy of CT was ∼80% for LG and 55% for SG. CONCLUSIONS: Ultrasonographic findings in IgG4-DS included diffuse or nodular low-echoic areas with linear high-echoic structures corresponding to inflamed lobules and inter-lobular fibrosis. These findings can help detect IgG4-DS.

Quantitative assessment of the parotid gland using computed tomography texture analysis to detect parotid sialadenitis.

OBJECTIVE: We aimed to quantitatively assess the parotid gland by using computed tomography (CT) texture analysis to detect parotid sialadenitis (PS). STUDY DESIGN: This retrospective case-control study included 43 patients with PS who underwent CT and magnetic resonance imaging (MRI). Parotid glands with an abnormal signal (STIR: High) on MRI were identified as showing PS. Patients with parotid gland tumors, bilateral PS, marked fatty degeneration, and severe artifacts on CT were excluded. The texture features of parotid glands with PS and the contralateral normal parotid glands were analyzed using the open-access software LIFEx. The regions of interest were manually placed by tracing contours of both parotid glands on CT images. The results were tested with the paired t-test (or Wilcoxon rank-sum test when appropriate). Receiver operating characteristic (ROC) curve analysis was performed to assess the ability of texture features to predict PS. RESULTS: Six gray level run length matrix features, 2 neighborhood gray level difference matrix features, and 5 gray level zone length matrix features displayed significant differences between PS and normal glands (P ≤ .047). ROC curve analysis showed acceptable accuracy in 4 texture features. CONCLUSIONS: CT texture analysis allowed quantitative assessment of parotid glands and may have the potential to detect PS.

[Acute sialadenitis by iodine contrast medium administration].

Acute iodide sialadenitis is a rare adverse reaction to iodinated contrast that causes self-limited salivary gland swell-ing. Its pathogenesis is still unclear, although kidney failure may be a risk factor. The diagnosis is initially clinical but angioedema, infections and lithiasis should be included in the differential diagnosis. No treatment or prophylaxis was proven to be beneficial. Although its prognosis is benign, associated complications have been reported. We report a case of 68-year-old man with swelling of the submandibular salivary glands after the administration of iodine-based contrast media during an abdominal computed tomography examination. Because of the widespread use of iodinated contrast enhanced imaging and interventional techniques, clinicians should be aware of this issue.

Standardization of terminology, imaging features, and interpretation of CBCT sialography of major salivary glands: a clinical review.

Sialography combined with cone beam computerized tomography (sialo-CBCT) is an imaging technique that demonstrates the ductal system of the major salivary glands and allows evaluation of gland function. This review describes the sialo-CBCT technique, terminology, common pitfalls and limitations, as well as radiographic features and suggested pathogenicity of various salivary gland disorders, based on 1,758 sialo-CBCT examinations conducted over the last decade in one institution, and the current literature. The adoption of standardized terminology is proposed to prevent miscommunication, facilitate formulation of differential diagnoses, and thereby promote patient management: (1) Sialo-CBCT requires specific training, and operator experience is required for adequate glandular filling with minimal extravasation; (2) Limit injection-to-scan time to avoid pre-mature emptying; (3) The sialo-CBCT report should include a description of the morphology of the primary duct as well as the secondary, tertiary, and descending branches, the maximal branching level, the presence of sialectasis, overall glandular size, and parenchymal findings; (4) Functional evaluation is based on assessment of iodine clearance in the post evacuation image; (5) Sialectasis and ductopenia are the main findings in Sjogren syndrome and recurrent juvenile parotitis; (6) Sialodochitis with or without fillings defects or hyperdense calcifications characterize obstructive sialadenitis and sialolithiasis; (7) The findings following radioactive-iodine-induced damage are similar to obstructive sialadenitis, with atrophy in late stages; (8) In chronic graft-versus-host disease (cGVHD), variable presentations of ductopenia, sialectasis, and sialodochitis may be evident; (9) The red flags indicating a space-occupying lesion include areas of no filling, splaying of ducts, and primary duct deviation.

Sialodochitis fibrinosa managed with staged bilateral total parotidectomy.

OBJECTIVE: This paper reports a rare case of a 61-year-old man with sialodochitis fibrinosa. METHODS: Clinical case report and review of current literature. RESULTS: Sialodochitis fibrinosa is a diagnosis of exclusion and in many cases can be managed conservatively. Conservative management failed for this patient and he was managed successfully with staged bilateral total parotidectomy. CONCLUSION: Sialodochitis fibrinosa should be considered as a differential diagnosis of painful bilateral facial swelling. While conservative management is successful for many patients, staged bilateral total parotidectomy may be necessary for full remission of symptoms; the timing of this is crucial to reduce the risk of facial nerve palsy.

Differential diagnosis of immunoglobulin G4-related sialadenitis and Kimura's disease of the salivary gland: a comparative case series.

The aim of this study was to investigate key points for the differential diagnosis of immunoglobulin G4-related sialadenitis (IgG4-RS) and Kimura's disease (KD) involving the salivary glands. The clinical, serological, radiological, histological, and immunohistochemical features of 85 IgG4-RS cases and 52 KD cases were evaluated comparatively. Seventy-two IgG4-RS cases had enlargement of multiple salivary and/or lacrimal glands; 67 patients had bilateral submandibular gland (SMG) involvement. Unilateral parotid gland involvement (59.6%) and comorbid skin lesions (61.5%) were common in KD. Serum IgG4 was elevated in 94.1% of IgG4-RS cases versus 19.0% of KD cases (cut-off value=266.5mg/dl). KD was more commonly associated with elevated eosinophil counts (86% vs 23.1%) and elevated IgE concentrations (95.5% vs 76.6%). Storiform fibrosis, irregular lymphoid follicles, and increased IgG4-positive cells (112.9±37.6/high-power field (HPF)) were common in IgG4-RS. Acellular fibrosis, regular lymphoid follicles, IgE-positive reticular networks, increased IgE-positive cells (43.4±26.7/HPF), and tryptase-positive mast cells (29.7±13.3/HPF) were usually detected in KD. Computed tomography showed that 85.7% of KD cases involved subcutaneous fat tissue. A superficial hypoechoic and reticular pattern with multiple hypoechoic foci were the sonographic features of the SMG in IgG4-RS. Despite numerous overlapping manifestations, histopathological examination showed meaningful differences in the types of fibrosis, eosinophils, and IgG4-positive cell counts. Comprehensive evaluation of clinical, serological, radiological, and histopathological features are crucial for the differential diagnosis.