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.

Clinical analysis of patients with oral and maxillofacial infections in oral emergency / 北京大学学报(医学版)

OBJECTIVE@#To analyze the composition, incidence and clinical characteristics of oral and maxillofacial infections in oral emergency.@*METHODS@#A retrospective study on patients with oral and maxillofacial infections who visited the Department of Oral Emergency in Peking University School and Hospital of Stomatology from January 2017 to December 2019 was conducted. General characteristics, such as disease composition, gender, age distribution and position of involved teeth were analyzed.@*RESULTS@#A total of 8 277 patients with oral and maxillofacial infections were finally collected, including 4 378 male patients (52.9%) and 3 899 female patients (47.1%), with gender ratio of 1.12:1. The common diseases were periodontal abscess (3 826 cases, 46.2%), alveolar abscess (3 537 cases, 42.7%), maxillofacial space infection (740 cases, 9.0%), sialadenitis (108 cases, 1.3%), furuncle & carbuncle (56 cases, 0.7%) and osteomyelitis (10 cases, 0.1%). Male patients were more easily affected by periodontal abscess, space infection and furuncle & carbuncle than female patients with the gender ratios 1.24:1, 1.26:1, 2.50:1 individually, while the incidence of alveolar abscess, sialadenitis, furuncle & carbuncle had no significant gender difference. Different diseases were prone to occur at different ages. The peak ages of alveolar abscess were 5-9 and 27-67 years, while the peak age of periodontal abscess was 30-64 years. Space infection tended to occur between 21-67 years. There were 7 363 patients with oral abscess (3 826 patients with periodontal abscess and 3 537 patients with alveolar abscess), accounting for 88.9% of all the patients with oral and maxillofacial infections, involving 7 999 teeth, including 717 deciduous teeth and 7 282 permanent teeth. Periodontal abscess usually occurred in permanent teeth, especially the molar teeth. Alveolar abscess may occur in both primary teeth and permanent teeth. In primary teeth, the most vulnerable sites were primary molar teeth and maxillary central incisors while in permanent teeth the most vulnerable sites were first molar teeth.@*CONCLUSION@#Understanding the incidence of oral and maxillofacial infection was conducive to the correct diagnosis and effective treatment of clinical diseases, as well as targeted education for patients of different ages and genders to prevent the occurrence of diseases.

Sialolitiasis, sialoadenitis y sialoadenosis. Revisión de literatura y reporte de un caso clínico / Sialolithiasis, sialadenitis and sialadenosis. Review of literature and report of a clinical case

Introducción: la formación de sialolitos se considera la alteración más común en las glándulas salivales. Su origen aún no es claro en cuanto a qué provoca la obstrucción parcial o total del sistema de conductos salivales, con una predilección significativa en las glán - dulas submandibulares, lo que permite un desplazamiento retrógrado de bacterias a través del conducto salival hacia la glándula afectada, el cual se traducirá clínicamente como inflamación unilateral o bila - teral. El tratamiento para la sialolitiasis y sialoadenitis puede variar desde un manejo conservador hasta tratamientos que implican la remoción de la glándula salival afectada. La sialoadenosis difiere de las anteriores en su origen, ya que ésta no se considera inflamatoria ni neoplásica y no afecta la función glandular de excretar saliva. Presentación de caso clínico: se trata de paciente masculino con diagnósticos presuntivos de sialoadenitis y sialolitiasis, al cual se le realizó biopsia escisional de la glándula afectada, su estudio histo- patológico corroboró los diagnósticos mencionados anteriormente que además confirmó un diagnóstico de sialoadenosis. Conclusio- nes: la escisión de la glándula salival involucrada se realizará en casos diagnosticados de sialoadenitis y sialolitiasis con sialolitos de gran tamaño y procesos supurativos crónicos que no resuelven con antibioticoterapia (AU)

Sialoendoscopia y abordaje mixto para el manejo dela patología obstructiva de las glándulas salivales / Sialoendoscopy and mixed approach for the management of obstructive pathology of the salivary glands

Introducción. La sialoendoscopia es un procedimiento cuya finalidad es visualizar los conductos salivales. Se utiliza como método diagnóstico y terapéutico de procesos inflamatorios, estenosis de los conductos y procesos obstructivos. Métodos. Describir los procedimientos realizados para el tratamiento de pacientes con patología inflamatoria y obstructiva de las glándulas salivales, de forma única con sialoendoscopia o con abordajes mixtos. Resultados. Un total de 24 pacientes fueron incluidos en el estudio, con edad promedio de 42 años, en su mayoría mujeres y compromiso submaxilar en 58,3 % y de parótida en 41,7 %. Respecto a la intervención, al 29,2 % de los sujetos se le realizó extracción de cálculos, al 29,2 % sialoplastia, al 25 % dilatación de conductos y al 37,5 % lavado de conductos en el mismo momento quirúrgico. Conclusión. La sialoendoscopia y el abordaje mixto es un procedimiento que puede garantizar el manejo de patologías obstructivas y estenosis de los conductos salivales, con buen pronóstico y resultados, preservando la glándula y evitando las complicaciones de la cirugía.

Introduction. Sialoendoscopy is a procedure which purpose is to visualise the salivary ducts. It is used as a diagnostic and therapeutic method for inflammatory and obstructive processes and duct stenosis. Methods. To describe the procedures performed for the management of patients with inflammatory and obstructive pathology of the salivary glands, only with sialoendoscopy or with mixed approaches. Results. A total of 24 patients were included in the study, with mean age of 42 years, mostly female, and 58.3% submaxillary involvement 41.7% parotid involvement. Regarding the intervention, 29.2% of the subjects underwent stone extraction, 29.2%, sialoplasty, 25% duct dilatation, and 37.5% duct lavage at the same surgical time. Conclusions. Sialoendoscopy and the mixed approach is a procedure that can guarantee the management of obstructive pathologies and stenosis of the salivary ducts with good prognosis and results, preserving the gland and avoiding the complications of surgery.

Sialoadenitis esclerosante asociada a enfermedad por IgG4: reporte de un caso / Sclerosing sialadenitis associated with IgG4 disease: case report

Las enfermedades relacionadas con IgG4 (ER-IgG4) son entidades fibroinflamatorias e inmunomediadas, caracterizadas por la afección multiorgánica, con la formación de pseudotumores que provocan lesión tisular y daño orgánico subsecuente. Se describe el caso de un paciente de 43 años que presentó sialoadenitis esclerosante y cumplió todos los criterios diagnósticos de enfermedad relacionada con IgG4.

IgG4-related diseases (IgG4-RD) are fibroinflammatory immune-mediated entities characterized by multiorgan involvement with the development of pseudotumors that cause tissue injury and subsequent organ damage. We describe the case of a 43-year-old man who presented sclerosing sialadenitis and fulfilled the diagnostic criteria for IgG4-related disease.

Interleukin-13 promotes cellular senescence through inducing mitochondrial dysfunction in IgG4-related sialadenitis / 国际口腔科学杂志·英文版

Immunoglobulin G4-related sialadenitis (IgG4-RS) is an immune-mediated fibro-inflammatory disease and the pathogenesis is still not fully understood. The aim of this study was to explore the role and mechanism of interleukin-13 (IL-13) in the cellular senescence during the progress of IgG4-RS. We found that the expression of IL-13 and IL-13 receptor α1 (IL-13Rα1) as well as the number of senescent cells were significantly higher in the submandibular glands (SMGs) of IgG4-RS patients. IL-13 directly induced senescence as shown by the elevated activity of senescence-associated β-galactosidase (SA-β-gal), the decreased cell proliferation, and the upregulation of senescence markers (p53 and p16) and senescence-associated secretory phenotype (SASP) factors (IL-1β and IL-6) in SMG-C6 cells. Mechanistically, IL-13 increased the level of phosphorylated signal transducer and activator of transcription 6 (p-STAT6) and mitochondrial-reactive oxygen species (mtROS), while decreased the mitochondrial membrane potential, ATP level, and the expression and activity of superoxide dismutase 2 (SOD2). Notably, the IL-13-induced cellular senescence and mitochondrial dysfunction could be inhibited by pretreatment with either STAT6 inhibitor AS1517499 or mitochondria-targeted ROS scavenger MitoTEMPO. Moreover, IL-13 increased the interaction between p-STAT6 and cAMP-response element binding protein (CREB)-binding protein (CBP) and decreased the transcriptional activity of CREB on SOD2. Taken together, our findings revealed a critical role of IL-13 in the induction of salivary gland epithelial cell senescence through the elevated mitochondrial oxidative stress in a STAT6-CREB-SOD2-dependent pathway in IgG4-RS.

Studies on newly recognized chronic sialadenitis / 北京大学学报(医学版)

Some kinds of chronic sialadenitis were recognized during the recent years. They have specific pathogenesis, clinical and histopathologic appearances, and require specific treatment. IgG4-related sialadenitis (IgG4-RS) is one of the immune-mediated diseases, characterized by tumefactive lesions. The incidence of IgG4-RS obviously increased during the past 30 years. The study on the potential relationship between occupational exposure to chemical substances and the incidence of IgG4-RS showed that subjects with occupational exposure to agents known to cause IgG4-RD had an increased risk for IgG4-RS. Surgical excision of involved SMG could not control the disease progression, which is not recommended for treatment of IgG4-RS. The combination of glucocorticoid and steroid-sparing agents is effective for treating IgG4-RS, and restores salivary gland function. Radioiodine induced sialadenitis (RAIS) is one of the common complications of postoperative adjuvant treatment of differentiated thyroid cancer by 131I. The incidence of the disease is related to radiation dosage. Clinically, the patients suffered from swelling and tenderness in the buccal or submandibular regions, especially during the mealtime. Imaging appearances are similar to those of chronic obstructive sialadenitis. Conservative managements, such as gland massage, sialagogues, are the mainstream methods in the treatment of RAIS. Sialendoscopy is feasible for RAIS, but not as effective as conventional obstructive sialadenitis (COS). Therefore the prevention of RAIS is crucial. Eosinophilic sialodochitis (ES) is a new type of chronic inflammatory disease of the salivary gland related to allergy. It has characteristics of swelling of multiple major salivary glands, strip-like gelatinous plugs discharged from the duct orifice of the gland, elevated level of serum IgE and eosinophils in peripheral blood, infiltration of eosinophils and IgE positive plasma cells in the tissues, allergic history, increased expression of allergy-related cytokines, such as IL-4, IL-5, IL-13, and eotaxin, which suggest allergic reactions as a potential pathogenesis of the disease. The clinical, laboratory, histological, and immunohistochemical characteristics of ES are significantly different from conventional obstructive sialadenitis (COS). Therefore, it is suitable to separate ES from COS. Conservative managements, such as self-maintenance therapy and anti- allergic modality are the choices of treatment for ES. Based on the results of our comprehensive studies a new classification of chronic sialadenitis is suggested.

Concordancia diagnóstica del sistema de graduación Chisholm Mason y Focus Score en la sialoadenitis linfocítica focal como aproximación al síndrome de Sjögren / Diagnostic agreement of the Chisholm Mason and Focus Score grading system in focal lymphocytic sialadenitis in patients with dry symptoms

RESUMEN Introducción: Existen diferentes sistemas de graduación para interpretar la biopsia de glándula salival menor (BGSM) en pacientes con síntomas secos. En la actualidad la metodología recomendada (Focus Score [FS]), aunque no se emplea de forma rutinaria, conlleva discrepancias diagnósticas. Objetivo: Determinar la concordancia de la lectura histopatológica de BGSM entre las técnicas FS y Chisholm Mason (CM) para el diagnóstico de sialoadenitis linfocítica focal en el hospital de San José, entre enero de 2017 y junio de 2018. Métodos: Estudio de concordancia. Se incluyeron BGSM de pacientes con síntomas secos. Se excluyó material no apto para el estudio, o con menos de 4 lóbulos o menos de 8 mm de área. Se clasificaron utilizando las técnicas FS y CM por 2 evaluadores independientes. Se evaluó el acuerdo interobservador, intraobsevador y global entre las pruebas mediante el coeficiente de Kappa de Cohen. Resultados: Se evaluaron 130 biopsias. Los valores K intraobservador entre FS y CM fueron 0,42 para el observador 1 y 0,23 para el observador 2. Los valores K del acuerdo interobservador fueron 0,47 para FS y 0,65 para CM. La concordancia entre los 2 sistemas de puntuación fue K de 0,13. Conclusiones: Se evidencia una probable sobreestimación de focos con CM, que resalta sus debilidades, y mayor precisión con FS. Por ende, la concordancia entre las 2 pruebas es débil. Es necesario un mayor esfuerzo para el uso del método estandarizado de lectura de BGSM con el FS para una correcta interpretación y beneficio en la clasificación de pacientes con síndrome seco.

ABSTRACT Introduction: There are different grading systems for interpreting the minor salivary gland biopsy (MSGB) in patients with dry symptoms (Sicca). The recommended methodology is currently the Focus Score (FS), which, although not performed routinely, results in diagnostic discrepancies. Objective: To determine the concordance of the histopathological reading of MSGB between the FS and Chisholm Mason (CM) techniques for diagnosis of focal lymphocytic sialadenitis at the San José Hospital between January 2017 and June 2018. Methods: Concordance study. MSGB of patients with sicca symptoms were included. Material not suitable for study and / or with less than 4 lobules and / or less than 8 mm of surface area was excluded. They were classified, using the FS and CM techniques, by two independent evaluators. The inter-observer, intra-observer, and overall agreement between the tests were evaluated using Cohen's Kappa coefficient. Results: A total of 130 biopsies were evaluated. The intra-observer K values between FS and CM were 0.42 for observer 1, and 0.23 for observer 2. The K values of the inter-observer agreement were 0.47 for FS, and 0.65 for CM. The concordance between the two scoring systems was K = 0.13. Conclusions: There is evidence of a probable over-estimation of foci with CM, highlighting its weaknesses and greater precision with FS technique, resulting in the weak concordance between the two tests. A greater effort is necessary for the use of the standardised MSGB FS reading method for a correct interpretation and benefit in the classification of patients with dry syndrome.

Sialolitectomía canicular por sialolito de gran tamaño / Canicular sialolithectomy of a giant sialolith

RESUMEN Introducción: La sialolitiasis es una afección frecuente de las glándulas salivales mayores que se manifiesta con mayor frecuencia en pacientes de sexo masculino. Objetivo: Presentar un caso clínico de una sialoadenitis por sialolitiasis de glándula submandibular, tratado exitosamente mediante sialolitectomía canicular intraoral. Caso clínico: Paciente de 45 años de edad atendido en el Departamento de Cirugía Maxilofacial de la Clínica de Especialidades "Manuel de Jesús Cedeño", Granma, Cuba, por presentar molestias en el suelo de la boca con aumento de volumen de consistencia dura, móvil y fácilmente desplazable. Radiográficamente, se observó una imagen radiopaca de bordes definidos, ovoide de aproximadamente 4 cm de largo y 2 cm de ancho. Se comprobó que se trataba de una sialoadenitis por sialolitiasis de la glándula submandibular derecha. Se trató la sintomatología mediante analgésicos-antiinflamatorios y antibióticos. Se realizó sialolitectomía canicular intraoral. Posteriormente se efectuó una recanalización del conducto lo que permitió la permeabilización de este. Conclusiones: La sialoadenitis por sialolitiasis es una afección con pronóstico favorable, pues cuando se realiza el tratamiento adecuado se logra que la glándula vuelva a su fisiología normal, lo que mejora la calidad de vida del paciente. En nuestro caso se realizó la remoción quirúrgica a través de sialolitectomía canicular intraoral, sin complicaciones. El paciente mostró excelente recuperación tras un mes de evolución, constituyendo un tratamiento satisfactorio a corto y largo plazo(AU)

ABSTRACT Introduction: Sialolithiasis is a common disorder of the major salivary glands which presents more frequently in male patients. Objective: Present a clinical case of sialadenitis due to sialolithiasis of the submandibular gland successfully treated by intraoral canicular sialolithectomy. Case report: A male 45-year-old patient attending the Maxillofacial Surgery Department at Manuel de Jesús Cedeño Specialty Clinic in Granma, Cuba, presents with discomfort on the floor of the mouth and a hard, mobile and easily movable mass of increased volume. Radiographic examination revealed a radiopaque oval-shaped image of well-defined edges, approximately 4 cm long x 2 cm wide. A diagnosis was made of sialadenitis due to sialolithiasis of the right submandibular gland. Symptoms were treated with analgesics / anti-inflammatories and antibiotics. Intraoral canicular sialolithectomy was performed. Rechanneling of the duct was then conducted to allow permeabilization. Conclusions: Sialadenitis due to sialolithiasis is a condition of a favorable prognosis. Appropriate treatment will have the gland return to its normal physiology, improving the patient's quality of life. In our case, surgical removal was conducted by intraoral canicular sialolithectomy without any complications. The patient had completely recovered after one month's evolution, the treatment being satisfactory both short- and long-term(AU)

Salivary Gland Disorders in Children and Adolescents: A 15-year Experience

Abstract Introduction Diseases of the salivary glands are rare in children and adolescents, with the exception of viral-induced infections. Objective To determine the clinical course of the disease, the diagnostic procedures, the treatment and the outcome of all children and adolescents affected with salivary gland diseases at our clinic over a period of 15 years. Methods A retrospective chart review including a long-term follow-up was conducted among 146 children and adolescents treated for salivary gland disorders from 2002 to 2016. Results Diagnosing acute sialadenitis was easily managed by all doctors regardless of their specialty. The diagnosis of sialolithiasis was rapidly made only by otorhinolar- yngologists, whereas diagnosing juvenile recurrent parotitis imposed difficulties to doctors of all specialties - resulting in a significant delay between the first occurrence of symptoms and the correct diagnosis. The severity-adjusted treatment yielded improve- ments in all cases, and a full recovery of 75% of the cases of sialolithiasis, 73% of the cases of juvenile recurrent parotitis, and 100% of the cases of acute sialadenitis. Conclusions Due to their low prevalence and the lack of pathognomonic symptoms, salivary gland diseases in children and adolescents are often misdiagnosed, resulting in an unneces- sarily long period of suffering despite a favorable outcome following the correct treatment.

Ultrasonography Echotexture as a surrogate for Sialadenitis secondary to 131I Radioiodine Therapy for differentiated Thyroid Cancer: a review and metaanalysis

To systematically review and analyze the medical literature to assess ultrasonography echotexture changes in thyroid cancer patients for the detection of chronic sialadenitis caused by radioiodine therapy. Methods: Sources were retrieved from PubMed, Scopus, EMBASE and LILACS through November 2018. All studies that assessed ultrasonographic features before 131I administration and at 12 months after 131I administration were selected. After data extraction, statistical analysis was performed by using Stata software. Results: From a total of 435 studies, 4 studies involving 665 patients were considered eligible, and echotexture heterogeneity was found with a significant difference. Conclusions: Ultrasound echotexture may detect chronic sialadenitis secondary to salivary radioiodine therapy.

Review of a novel disease entity, immunoglobulin G4-related disease

Immunoglobulin G4 (IgG4)-related dacryoadenitis and sialoadenitis (IgG4-DS) are part of a multiorgan fibroinflammatory condition of unknown etiology termed IgG4-related disease (IgG4-RD), which has been recognized as a single diagnostic entity for less than 15 years. Histopathologic examination is critical for diagnosis of IgG4-RD. CD4+ T and B cells, including IgG4-expressing plasma cells, constitute the major inflammatory cell populations in IgG4-RD and are thought to cause organ damage and tissue fibrosis. Patients with IgG4-RD who have active, untreated disease exhibit significant increase of IgG4-secreting plasmablasts in the blood. Considerable insight into the immunologic mechanisms of IgG4-RD has been achieved in the last decade using novel molecular biology approaches, including next-generation and single-cell RNA sequencing. Exploring the interactions between CD4+ T cells and B lineage cells is critical for understanding the pathophysiology of IgG4-RD. Establishment of pathogenic T cell clones and identification of antigens specific to these clones constitutes the first steps in determining the pathogenesis of the disease. Herein, the clinical features and mechanistic insights regarding pathogenesis of IgG4-RD were reviewed.

Inflammation grading and sialoendoscopic treatment of 131I radioiodine-induced sialadenitis / 北京大学学报(医学版)

OBJECTIVE@#To investigate the inflammation grading of 131I radioiodine-induced sialadenitis based upon sialoendoscopic and sialographic appearances, and to evaluate the results of sialoendoscopic intervention.@*METHODS@#The patients diagnosed with 131I radioiodine-induced sialadenitis and underwent sialoendoscopic exploration and intervention procedures in Peking University Hospital of Stomatology from Nov. 2012 to Oct. 2018 were included in this study. The appearances of sialogaphy and sialoendoscopy were analyzed and classified. The treatment options included irrigation with saline and dexamethasone and mechanical dilatation by sialoendoscope. The patients were followed up after treatment.@*RESULTS@#Forty-two patients with 131I radioiodine-induced sialadenitis were included. There were 5 males and 37 females, with a male-to-female ratio of 1 ∶7.4. Symptoms included recurrent swelling and pain in the parotid glands, and dry mouth. Sialography showed stenosis in the main duct,and in some cases nonvisua-lization of the branches. Sialoendoscopy showed narrowing of the main duct, and the branch duct atresia was seen. The appearances of sialogaphy and sialoendoscopy were analyzed and classified into 3 groups: (1) Mild inflammation: stenosis and ectasia occurred in the main duct, whereas the 0.9 mm sialoendoscope could pass through easily. (2) Moderate inflammation: one point of severe stricture could be seen in the main duct where 0.9 mm sialoendoscope could not be passed through. (3) Severe inflammation: two points or more of severe strictures or diffused strictures occurred in the main duct. Thirty-three patients with 65 affected glands were examined by both sialography and sialoendoscopy. Eight glands were classified as mild inflammation, 23 glands moderate inflammation, and 34 glands severe inflammation. The duration of follow-up ranged from 3-72 months. The clinical results were evaluated as good in 22 glands, fair in 22 glands, and poor in 19 glands, with an overall effective rate of 69.8% (44/63).@*CONCLUSION@#The clinical, sialographic and sialoendoscopic appearances of 131I radioiodine-induced sialadenitis showed their characteristics. We proposed an inflammation grading standard for the 131I radioiodine-induced sialadenitis based on the appearances of sialography and sialoendoscopy. Sialoendoscopy can significantly alleviate the clinical symptoms, which is an effective therapy, and better for early lesions.

Sialoadenite esclerosante crônica em glândula salivar menor / Chronic sclerosing sialoadenitis in small salivary gland

A sialoadenite esclerosante crônica, também conhecida como tumor de Kutner, é uma condição, que acomete, predominantemente, glândulas salivares maiores. Clinicamente, apresenta-se como um nódulo, móvel, assintomático e de consistência endurecida. Seu diagnóstico definitivo só é conseguido através da análise histopatológica onde estão presentes: intenso infiltrado inflamatório linfocítico, atrofia acinar, fibrose do parênquima glandular e graus variáveis de esclerose. Neste artigo, é relatado um caso clínico raro de sialoadenite esclerosante crônica, localizada em glândula salivar menor... (AU)

Chronic sclerosing sialoadenitis, also known as Kutner's tumor, is a condition that predominantly affects major salivary glands. Clinically it presents as a nodule, mobile, asymptomatic, and of hardened consistency. Its definitive diagnosis is only achieved through the histopathological analysis where they are present: intense lymphocytic inflammatory infiltrate, acinar atrophy, fibrosis of the glandular parenchyma and variable degrees of sclerosis. In this article we report a rare clinical case of chronic sclerosing sialoadenitis located in the minor salivary gland... (AU)

Surgical Treatment of Chronic Parotitis

Abstract Introduction chronic parotitis (CP) is a hindering, recurring inflammatory ailment that eventually leads to the destruction of the parotid gland. When conservative measures and sialendoscopy fail, parotidectomy can be indicated. Objective to evaluate the efficacy and safety of parotidectomy as a treatment for CP unresponsive to conservative therapy, and to compare superficial and near-total parotidectomy (SP and NTP). Methods retrospective consecutive case series of patients who underwent parotidectomy for CP between January 1999 and May 2012. The primary outcome variables were recurrence, patient contentment, transient and permanent facial nerve palsy and Frey syndrome. The categorical variables were analyzed using the two-sided Fisher exact test. Alongside, an elaborate review of the current literature was conducted. Results a total of 46 parotidectomies were performed on 37 patients with CP. Neartotal parotidectomy was performed in 41 and SP in 5 cases. Eighty-four percent of patients was available for the telephone questionnaire (31 patients, 40 parotidectomies) with a mean follow-up period of 6,2 years. Treatment was successful in 40/46 parotidectomies (87%) and 95% of the patients were content with the result. The incidence of permanent and transient facial nerve palsy was 0 (0%) and 12 (26.1%), respectively. Frey syndrome manifested in 20 (43.5%) patients. Neither this study nor careful review of the current literature resulted in evident difference between SP and NTP regarding the primary outcome variables. Conclusion parotidectomy is a safe and effective treatment for CP in case conservative therapy fails. There is no evidence of a distinct difference between SP and NTP regarding efficiency, facial nerve palsy or Frey syndrome. (AU)

Aged Sanroque Mice Spontaneously Develop Sjögren's Syndrome-like Disease

Sjögren's syndrome (SS) is a chronic inflammatory autoimmune disorder that affects mainly salivary and lacrimal glands, but its cause remains largely unknown. Clinical data indicating that SS occurs in a substantial proportion of patients with lupus points to common pathogenic mechanisms underlying the two diseases. To address this idea, we asked whether SS develops in the lupus-prone mouse strain sanroque (SAN). Owing to hyper-activation of follicular helper T (Tfh) cells, female SAN mice developed lupus-like symptoms at approximately 20 wk of age but there were no signs of SS at that time. However, symptoms typical of SS were evident at approximately 40 wk of age, as judged by reduced saliva flow rate, sialadenitis, and IgG deposits in the salivary glands. Increases in serum titers of SS-related autoantibodies and numbers of autoantibody-secreting cells in cervical lymph nodes (LNs) preceded the pathologic manifestations of SS and were accompanied by expansion of Tfh cells and their downstream effector cells. Thus, our results suggest that chronic dysregulation of Tfh cells in salivary gland-draining LNs is sufficient to drive the development of SS in lupus-prone mice.

Primary Necrobiotic Xanthogranulomatous Sialadenitis with Submandibular Gland Localization without Skin Involvement

Necrobiotic xanthogranulomatous reaction is a multiorgan, non-Langerhans cell histiocytosis with an unknown etiology. Occurrence in the salivary gland is extremely rare. We recently identified a case of necrobiotic xanthogranulomatous sialadenitis in a 73-year-old Korean woman who presented with a painless palpable lesion in the chin. There was no accompanying cutaneous lesion. Partial resection and subsequent wide excision with neck dissection were performed. Pathological examination showed a severe inflammatory lesion that included foamy macrophages centrally admixed with neutrophils, eosinophils, lymphocytes, plasma cells, and scattered giant cells, as well as necrobiosis. During the 12-month postoperative period, no grossly remarkable change in size was noted. Necrobiotic xanthogranulomatous inflammation may be preceded by or combined with hematologic malignancy. Although rare, clinicians and radiologists should be aware that an adhesive necrobiotic xanthogranuloma in the salivary gland may present with a mass-like lesion. Further evaluation for hematologic disease and close follow-up are needed when a pathologic diagnosis is made.

Circulating CCR7(lo)PD-1(hi) Follicular Helper T Cells Indicate Disease Activity and Glandular Inflammation in Patients with Primary Sjögren's Syndrome

Since primary Sjögren's syndrome (pSS) is an autoummune disease of B cell hyperactivity and pathologic autoantibody response, follicular helper T (Tfh) cells and follicular regulatory T (Tfr) cells are suggested to be key players in pSS. We examined subsets of Tfh and Tfr cells from the blood in pSS patients, and whether these subsets represent disease activity, glandular inflammation, or autoantibody responses in pSS. Circulating Tfh and Tfr cells, along with their specific subsets, were identified from the peripheral blood of 18 pSS patients and 14 age- and sex-matched healthy controls (HCs) using flow cytometry analysis. Blood Tfr and Tfh cell ratios were increased in pSS patients compared with HCs. The CCR7(lo)PD-1(hi) subset of circulating Tfh cells was increased in pSS patients with high degree of focal lymphocytic sialadenitis; whereas circulating Tfh cells did not differ between pSS patients and HCs. The frequency of CCR7(lo)PD-1(hi) Tfh cells was significantly correlated with disease activity scores and differentiated B cells. PD-1 expression on blood Tfh and Tfr cells showed positive correlations with IL-21 in pSS. Increasing trend of blood Tfr cells was observed in pSS patients, and blood Tfr cells (particularly Th1 and Th17 subsets) represented hypergammaglobulinemia in pSS. In summary, circulating CCR7(lo)PD-1(hi) Tfh cells indicated disease activity and glandular inflammation in pSS. Circulating Tfr cells, shifted toward Th1 and Th17 subsets, indicated ongoing IgG production in pSS. Subsets of circulating Tfh or Tfr cells could be biomarkers for disease monitoring and patient stratification in pSS.

Quantitative Analysis of Parotid Gland Secretion Function in Sjögren's Syndrome Patients with Dynamic Magnetic Resonance Sialography

OBJECTIVE: To evaluate the secretory function of parotid glands by dynamic magnetic resonance (MR) sialography and determine the clinical performance of this technique in diagnosing and evaluating Sjögren's syndrome (SS) patients. MATERIALS AND METHODS: This study enrolled 29 healthy volunteers (25 women and 4 men; mean age, 34.8 ± 6.3 years; age range, 26–47 years) and 25 primary SS (pSS) patients (23 women and 2 men; mean age, 37.7 ± 7.9 years; age range, 25–50 years) with decreased secretory function. The volume of the parotid gland ducts was precisely measured for both groups at single pre- and 6 post-gustatory-stimulated phases. Time-dependent volume change ratio curves were generated, four parameters were derived from the curves: the slope of the increase in the first post-stimulation phase (slope(1st)), the peak value, the time-to-peak, the total saliva secretion post-stimulation. All values were used to quantitatively evaluate the secretory function of the parotid gland. The repeated measurement analysis, Mann-Whitney U test and receiver operating characteristic curve were applied. RESULTS: Time-dependent volume change ratio curves demonstrated that there is a statistically significant difference between the two groups (F = 8.750; p = 0.005). A quickly increasing curve was shown in the volunteer group, whereas a slowly increasing curve was shown in the pSS patient group. The slope(1st), peak value and total saliva secretion post-stimulation of the patient group were significantly lower than those of the volunteer group (p = 0.005, p = 0.003, and p = 0.002, respectively). The time-to-peak between the two groups was not significantly different (p = 0.383). The slope(1st) can be used as a discriminator to diagnose SS patients (p = 0.015; odds ratio = 4.234; area under the curve = 0.726). CONCLUSION: Dynamic MR sialography is proven to be an effective method in evaluating salivary gland function and has a great potential in diagnosing and evaluating pSS patients.

Application of ultrasonography scoring system in the assessment of IgG4-related sialadenitis / 北京大学学报(医学版)

OBJECTIVE@#To assess the diagnostic value of salivary gland ultrasonography (SGUS) for IgG4-related sialadenitis.@*METHODS@#Ultrasonography examination of major salivary glands was conducted for 48 IgG4-related sialadenitis patients and 50 Sjögren's syndrome patients, whose ages and disease duration were matched. The imaging features were graded using two different scoring systems (0-16 and 0-48, respectively) obtained from the grades of bilateral parotid and submandibular glands. The scores were used to further evaluate the features of salivary gland ultrasonography in IgG4-related sialadenitis and to compare them with Sjögren's syndrome patients. The association of SGUS scores of IgG4-related sialadenitis group with serological tests was analyzed.@*RESULTS@#The mean age of IgG4-related sialadenitis group and Sjögren's syndrome group was 49.23 years and 50.44 years, respectively. The serum IgG4 level of the patients in the IgG4-related sialadenitis group was increased, with an average (9.60±6.43) g/L. And the serum IgE level was at a median of 251.5 (123.4-543.6) IU/mL. In the 0-16 system, the scores of submandibular glands of the patients in IgG4-related sialadenitis and Sjögren's syndrome were 6.0 (6.0-8.0) and 4.0 (2.0-8.0), and the scores of the total four glands were 10.0 (8.0-14.0) and 8.0 (4.0-12.0) respectively. In the 0-48 system, the scores of submandibular glands with IgG4-related sialadenitis and Sjögren's syndrome were 18.0 (14.5-20.0) and 11.0 (7.0-14.0), and the scores of the total four glands were 26.0 (18.5-34.0) and 21.5 (15.0-26.3) respectively. It suggested that in the 0-16 system and the 0-48 system, scores of submandibular glands and the total of four glands of IgG4-related sialadenitis were higher than those of Sjögren's syndrome. Meanwhile, the association analysis of 0-48 system showed a positive correlation of SGUS scores with serum IgG4, which also showed a positive correlation of SGUS scores with serum IgE in 0-16 system.@*CONCLUSION@#Semi-quantitative ultrasonography scoring systems can evaluate and quantify the lesions of salivary glands, which can be helpful in the diagnosis and differential diagnosis of IgG4-related sialadenitis combined with the clinical manifestations, serological indicators and/or histopathological manifestations. Ultrasonography can also assess the activity of IgG4-related sialadenitis preliminarily.