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SAT0500 JUVENILE RECURRENT PAROTITIS: A RARE MANIFESTATION OF RHEUMATIC IMMUNE-MEDIATED DISEASES

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Background: Juvenile recurrent parotitis (JRP) is the second most common childhood disease of the salivary glands after mumps and mainly affects children between the ages of 3 and 6. There… Click to show full abstract

Background: Juvenile recurrent parotitis (JRP) is the second most common childhood disease of the salivary glands after mumps and mainly affects children between the ages of 3 and 6. There is a male predilection, often with spontaneous resolution at puberty. However, in some cases, it can be the first manifestation of a rheumatic immune-mediated disease. Objectives: To analyze the clinical, laboratory and imaging profile of children with JRP and investigate the prevalence of rheumatic immune-mediated diseases in these patients. Methods: Retrospective study from 2008 to 2018 including all cases of recurrent parotitis with juvenile onset at our center. Parameters evaluated included gender, age, laterality, number of recurrences, symptoms of presentation, associated conditions, imaging details, blood tests, treatment, outcome and follow-up. Results: 40 patients were identified over a 10-year period, with a female to male ratio of 5:3; 62.5% females. The youngest child was nine months old and the eldest was 16 years old with median age at presentation of 5 years. The median (min-max) follow-up period was 3 (0.04-33) years. 29 children (72.5%) only ever reported unilateral symptoms, while the rest had both glands affected, although not usually at the same time. Pain and swelling were the most common presenting symptoms, seen in all cases. Fever in 15 (37.5%) and whitish discharge from Stenson’s duct in one (2.5%). 14 (35%) had palpaple cervical lymphadenopathy and 4 (10%) had dental caries. The average reported frequency was 1.25 episodes per year, with a range of 0.43-5 episodes. Numbers of recurrences were in range of one to 15 with average of 3 times. Younger age at first episode didn’t increase the likelihood of recurrences in our sample (p=0.123). Sonography was performed in 34 cases as primary investigation. Reports showed multiple hypoechoic lesions with enlarged intraglandular lymph nodes and enlarged gland. MRI was performed in 10, CT in 4, scintigraphy in 2 and biopsy of minor salivary glands in 1. The median level of C-reactive protein and erythrocyte sedimentation rate was 4 (range 0.2-55.5 mg/l, n=30) and 7 (0.5-76 mm in 1st hour, n=20), respectively. The median blood leukocyte count 8.13 x 109/L (range 3.73-25.4 x l09/L, n = 36). 20 (50%) patients had serum immunoglobulins measured, 6 of them having hypergammaglobulinaemia. One patient had a marginally low serum Immunoglobulin A (50; normal variation 55-210). Antibody screening was performed in 18 patients (45%), with 10 with positivity for antinuclear antibodies, 3 anti-SSA, 2 anti-SSB, 2 anti-RNP and 2 rheumatoid factor. One of the children had sialolithiasis in the parotid gland and other had an abcess with microaerophilic streptococcus isolated in culture of pus. Among all patients, 5 (12.5%) had a final diagnosis of a rheumatic immuno-mediated disease: 3 with primary Sjögren Syndrome (SS) and 2 with mixed connective tissue disease. An idiopathic etiology was determined in 33 (82.5%). These were treated with analgesics and nonsteroidal antiinflammatory drugs with complete resolution of symptoms and the previous ones with immunossupressants/immunomodulators. Conclusion: The clinical, laboratory and imaging profile of our sample is in accord with the literature, except for sex predilection. Although recurrent juvenile parotitis is more common, primary juvenile SS should be considered. A high suspicion is crutial mainly because of a lower incidence of xerostomy and xerophthalmia in this population. References [1] De Luca R, et al. Juvenile Recurrent Parotitis: Could be a Primary Pediatric Sjogren Syndrome? SM Otolaryngol. 2017; 1(3): 1013. Disclosure of Interests: None declared

Keywords: rheumatic immune; immune mediated; juvenile recurrent; parotitis; recurrent parotitis

Journal Title: Annals of the Rheumatic Diseases
Year Published: 2019

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