Confirmation of eosinophilic sialodochitis by terminal duct biopsy.

OBJECTIVE: To analyse the histopathological features of eosinophilic sialodochitis by using terminal duct biopsy. METHODS: Sixty-five patients with suspected eosinophilic sialodochitis and four with chronic obstructive sialadenitis were prospectively enrolled. Clinical features, laboratory tests and sialograms were comparatively analysed. Terminal duct biopsy of the parotid or submandibular glands was performed concomitantly with endoscopy-assisted duct dilatation to determine the histopathological features of eosinophilic sialodochitis. RESULTS: Based on eosinophil quantification, the samples of suspected patients were scored as 'definite', 'highly suspected' and 'negative' in 26 (40%), 15 (23.1%) and 24 (36.9%) cases, respectively. Gland types and peripheral blood eosinophil counts were significantly different among these three groups. The proportions of itching glands, mucus plug exudations and elevated immunoglobulin E levels were higher in the 'definite' group than in the other two groups; however, the intergroup differences were insignificant. The primary pathological features of eosinophilic sialodochitis were abundant eosinophils and lymphocytes infiltrated around the duct, degranulation of eosinophils, extensive fibrosis and scattered mastocytes. Periductal eosinophils were not found in cases of chronic obstructive sialadenitis. CONCLUSION: Our findings suggest that terminal duct biopsy is safe and valuable for the pathological confirmation of eosinophilic sialodochitis, and can be used simultaneously with endoscopy-assisted duct dilatation.

[Studies on newly recognized chronic sialadenitis].

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.

Eosinophilic sialodochitis: An emerging atopic condition.

OBJECTIVE: To report the clinical characteristics of the largest single centre cohort of patients with eosinophilic sialodochitis. METHODS: Analysis of data relating to 37 patients seen in a dedicated multidisciplinary clinic was performed. Demographic, clinical, haematological, cytological, histological and radiological features were collated. Response to trials of allergy treatment was assessed. RESULTS: Thirty-seven patients (30 female, seven male) were identified, 42% of whom were of Afro-Caribbean origin, with a mean age of 50.4 years (range 28-80 years). Mean symptom duration at presentation was 10 years (range 2-33 years). Parotid and submandibular gland involvement was equally reported. The most commonly reported symptoms were swelling (97%), itching of the overlying skin (92%), salivary gland discomfort (84%) and "string-like" mucus discharge from salivary duct orifices (76%). Twenty-three patients (62%) demonstrated atopic disease and serum IgE level elevated in 57%. All 37 patients had eosinophils present in aspirated duct contents samples while raised peripheral eosinophil count was seen in 41%. Anecdotal symptom improvement was reported with antihistamine, antileukotriene or steroid treatment. CONCLUSION: Eosinophilic sialodochitis should be considered in any patient presenting with recurrent salivary gland swelling. Further studies are needed to evaluate treatments directed at a likely allergic pathogenesis.

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.

Allergy-Related Sialodochitis: A Preliminary Cohort Study.

OBJECTIVES/HYPOTHESIS: To explore the clinically feasible diagnosis criteria and treatment outcomes of allergy-related sialodochitis (ARS). STUDY DESIGN: Prospective Cohort Study. METHODS: Ninety-six consecutive patients were enrolled by the following criteria: 1) recurrent swelling of ≥2 large salivary glands that lasted for ≥3 months; 2) with mucus plug exudations; 3) with atopic diseases; 4) ductal stenosis and/or ectasia. Sixty-four patients with elevation of peripheral blood eosinophil (PBE) and/or serum IgE level comprised group A (highly-suspected ARS group), while the remaining 32 comprised group B (patients without confirmed evidence of ARS). These patients were treated with interventional endoscopy. A chronic obstructive sialadenitis symptom (COSS) questionnaire was used to quantify the treatment outcomes. RESULTS: In group A, Serum IgE was elevated in 84.4% of patients and PBE was elevated in 34.4% of patients. Percentage of submandibular gland involvement was higher in group A than group B (48.4% vs. 18.8%). On sialograms, the snowflake changes of branch ducts were seen in higher percentage of group A compared with group B (59% vs. 35% for parotid glands, 27% vs. 8% for submandibular glands, respectively). Mucus plug smears showed abundant eosinophils in 14 group A patients. Biopsy of five group A patients revealed significant eosinophil infiltration around the main and interlobular ducts. During follow-up, the COSS scores were significantly decreased in both groups, and group B was improved better than group A. CONCLUSION: PBE and serum IgE are important diagnostic indexes of ARS. Mucus plug smear or histopathology verifies the diagnosis. Interventional endoscopy is helpful for ARS cases. LEVEL OF EVIDENCE: 3 Laryngoscope, 131:2030-2035, 2021.

Eosinophilic Sialodochitis: A Type of Chronic Obstructive Sialadenitis Related to Allergy.

OBJECTIVES: To investigate the clinical, laboratory, radiological, histopathological, and immunohistochemical features, and the expression of allergy-related cytokines in eosinophilic sialodochitis (ES). METHODS: Thirty-eight patients diagnosed with chronic obstructive sialadenitis (COS) who had undergone glandular excision or incisional biopsy were enrolled. Seventeen patients with comorbid atopic disease and increased ductal tissue eosinophils comprised the ES group, while 21 patients comprised the COS group. The clinicopathological features and allergy-related cytokine expression were compared between groups. RESULTS: The ES group frequently involved multiple, bilateral major salivary glands, and the number of glands was significantly greater than the COS group (2.8 ± 1.1 vs. 1.2 ± 0.4, P < .001). Serum immunoglobulin (Ig) E was elevated in 91% of patients in ES group (419 ± 357 kU/L) and peripheral blood eosinophil was significantly greater compared with the COS group (7.6% ± 4.6% vs. 2.5% ± 1.4%, P < .001). Histologically, eosinophil infiltration in ES group was observed around the main and interlobular ducts (50 ± 39/high power field [HPF]). Follicular hyperplasia (76%), epithelial mucous metaplasia (82%), and mucus plugs with eosinophils (41%) were observed. IgE-positive cell count was 20.7 ± 18.3/HPF and tryptase-positive mast cell count was 23.5 ± 15.1/HPF, which was significantly greater than the respective cell counts in COS group, which mainly infiltrated around the ducts. The levels of interleukin-4, interleukin-13, and eotaxin in tissue were significantly greater in ES than the COS group. CONCLUSIONS: The clinicopathological characteristics of ES are significantly different from COS and ES might have an allergy-related pathogenesis. LEVEL OF EVIDENCE: 4 Laryngoscope, 131:E800-E806, 2021.

The Positive Effect of Sialendoscopy with Irrigation Lavage for Sialadenitis without Sialolithiasis or Stenosis.

AIMS: The aim of the study was to evaluate different irrigation solutions during sialendoscopy to reduce complaints caused by sialodochitis. METHODS: The investigators designed and implemented a retrospective study composed of 94 patients with sialodochitis but no sialolithiasis or relevant stenosis of the salivary duct system. Three different irrigations (normal saline, cortisone single-shot, and cortisone) were used. After the sialendoscopy, the patients were asked about the current status of the affected salivary gland, and the quantity and quality of the gland swelling/inflammation over a follow-up period of 11.6 months (6-24 months). RESULTS: At the time of follow-up, 41 patients (43.6%) no longer had any complaints with the salivary gland in question. In 75.5% of the patients, a satisfying result was achieved. In total, only 3 patients needed submandibulectomy in the course of the study due to persistent complaints. The outcome showed no statistical differences between the mean of the 3 groups (p = 0.149). CONCLUSION: Sialendoscopy with flushing/lavage seems to be a good treatment option for patients with sialodochitis lacking sialolithiasis. The results of this study suggest that the type of irrigation used during sialendoscopy seems to be secondary.

Therapeutic effect of intraductal irrigation of the salivary gland: A technical report.

PURPOSE: Obstructive and inflammatory disease often occurs in the major salivary glands, and no predictive treatment has yet been developed for this condition. The aim of this report was to introduce an intraductal irrigation procedure and to illustrate its application to practical patient cases. MATERIALS AND METHODS: Two patients complaining of pain and swelling in the parotid gland during meals who underwent sialography were diagnosed as having sialodochitis with sialadenitis. Intraductal irrigation was then performed on the parotid gland on the side of the complaint. The irrigation procedure was conducted in the same manner as the sialography procedure, except that saline was used as the filling solution. Symptom severity was evaluated with a numerical rating scale (NRS) at the initial visit and a month after the irrigation. RESULTS: The initial NRS value of patient 1 was 10. The value decreased to 6 and then to 0 after 2 irrigation procedures. The NRS value of patient 2 regarding the symptoms involving the left parotid gland decreased from 4-5 to 1 after 4 irrigation procedures performed at 1-month intervals. CONCLUSION: Intraductal irrigation of the salivary gland may be a simple, safe, and effective treatment option for patients with obstructive and inflammatory disease of the salivary gland that is capable of resolving their symptoms.

Eosinophilic sialodochitis: redefinition of 'allergic parotitis' and 'sialodochitis fibrinosa'.

Sialodochitis fibrinosa and allergic parotitis have described rare patients with recurrent salivary gland swelling and mucus plugs, often with atopy. We have evaluated three patients with atopic disease, recurrent salivary gland swelling, and an eosinophilic sialodochitis. Two had eosinophil-rich mucus plugs. Fifty-six additional cases were identified in a medical literature database search, each defined by recurrent salivary gland swelling associated with eosinophil-rich mucus plugs or sialodochitis with periductal eosinophilic infiltration. The majority (78%) were reported from Japan. Females were predominantly affected (F:M = 2.3) with a median age of 47 years at evaluation. The parotid and submandibular glands were involved, respectively, in 71% and 46%. Allergic symptoms were present in 66%, atopic disease in 63% of those with reported allergy testing, and blood eosinophilia in 71%. Contrast sialography and other imaging modalities documented ductal dilatation in 82%. Treatments included anti-allergic medications (58%), systemic glucocorticoids (25%), duct cannulation with irrigation, steroid injection, and/or duct dilatation (36%), and glandular resection (19%). We recommend the diagnosis 'eosinophilic sialodochitis' be applied to patients who meet this case definition. The disease is a unique cause of chronic recurrent salivary gland swelling. Its likely allergic etiology may be amenable to current or future biologic therapies.

Therapeutic effect of intraductal irrigation of the salivary gland: A technical report

PURPOSE: Obstructive and inflammatory disease often occurs in the major salivary glands, and no predictive treatment has yet been developed for this condition. The aim of this report was to introduce an intraductal irrigation procedure and to illustrate its application to practical patient cases. MATERIALS AND METHODS: Two patients complaining of pain and swelling in the parotid gland during meals who underwent sialography were diagnosed as having sialodochitis with sialadenitis. Intraductal irrigation was then performed on the parotid gland on the side of the complaint. The irrigation procedure was conducted in the same manner as the sialography procedure, except that saline was used as the filling solution. Symptom severity was evaluated with a numerical rating scale (NRS) at the initial visit and a month after the irrigation. RESULTS: The initial NRS value of patient 1 was 10. The value decreased to 6 and then to 0 after 2 irrigation procedures. The NRS value of patient 2 regarding the symptoms involving the left parotid gland decreased from 4-5 to 1 after 4 irrigation procedures performed at 1-month intervals. CONCLUSION: Intraductal irrigation of the salivary gland may be a simple, safe, and effective treatment option for patients with obstructive and inflammatory disease of the salivary gland that is capable of resolving their symptoms.