Finding of celiac disease during the study of Kimura disease in a Caucasian male.

Abstract

We present the case of a 27-year-old man, with no previous diseases, who was referred to the Department of Otorhinolaryngology in May 2023 for a 2-cm right submandibular lymphadenopathy of two month's evolution. He did not report fever or night sweats, and no oral or genital ulcers were observed. He presented eosinophilia in the analysis performed and a maculopapular exanthema on the left leg and malleolus areas, which was initially treated with topical antibiotic and corticosteroids. A soft tissue ultrasound revealed two right laterocervical lymphadenopathies at levels I and II. Given the suspicion of a primary hematological tumor, a PET-CT scan was conducted to complete the study, which revealed hypermetabolic laterocervical bilateral lymphadenopaties. In June 2023, a programmed cervicotomy was conducted, with complete excision of the right main lymphadenopathy in the Ib region, which showed a histological result of reactive lymphadenitis with intense eosinophilia, vascular proliferation, and fibrosis. These findings suggest Kimura disease as the first possibility. Due to sporadic episodes of choking that were not associated with specific food, persistent elevated IgE levels and eosinophilia in peripheral blood tests, and skin alterations, the study was completed with a gastroscopy to rule out the presence of eosinophilic esophagitis, which showed erosive duodenitis, with villous shortening and increased lymphoplasmacytic cellularity of the lamina propria, findings consistent with celiac disease, which was afterwards confirmed with the presence of positive anti-endomysial and transglutaminase antibodies, and the presence of HLA-DQ8 positive in homozygosis in the genetic study. The patient started systemic treatment with corticosteroids and gluten-free diet, which he continues to take at present.