ESPE Abstracts

Background: Sjogren’s syndrome is a systemic autoimmune disease, and it is rare in pediatric population. Primary symptoms are dry mouth and dry eyes. However, it may present with systemic manifestations, involving lungs, liver, kidneys, vasculature, and blood. We report a case of Sjogren's syndrome with systemic involvement in a child with Graves' disease.

Case report: A 9-year-old 11-month-old girl presented to the emergency department with complaints of fever for 2 days. She was diagnosed with Graves' disease a year ago and has been treated with 10 mg/day of methimazole. Her laboratory finding showed leukopenia and mild elevation of pancreatic enzymes. And hyperthyroidism was exacerbated. On post admission day 4, fever continued, and neck swelling was observed. Ultrasonography on her neck revealed diffuse swelling of the both parotid glands with numerous tiny calcific foci, suggesting chronic sialadenitis such as autoimmune parotitis. Further evaluation was performed considering Sjogren’s syndrome as the cause of autoimmune parotitis. She complained of dry mouth and dry eyes, and ophthalmic examination showed abnormal Schirmer’s test. Laboratory finding showed positive Anti-Ro/SS-A antibody and antinuclear antibody. Bone marrow aspiration was unremarkable. Her salivary scan showed decreased wash-out in the bilateral parotid and submandibular glands with normal uptake, suggesting duct stricture or obstruction. She was diagnosed with Sjogren’s syndrome with systemic involvement, and prednisolone (2mg/kg daily) and pilocarpine was started. The fever subsided, and hyperthyroidism with improved normalized.

Conclusion: Systemic involvement of Sjogren's syndrome combined with hyperthyroidism in children is rare and glucocorticoid could be the choice of treatment. In patients with an autoimmune disease, the possibility of developing another autoimmune disease should be considered.)