Year 2026 / Volume 118 / Number 1
Letter
Finding of celiac disease during the study of Kimura disease in a Caucasian male

56-57

DOI: 10.17235/reed.2024.10853/2024

Elena Pablo-Martín, Laura Corvo-Félix, Marta Rodríguez González, Andrea Beatriz Jiménez Pérez, Luis Figuero-Pérez, María Luisa Pérez García, Emilio Fonseca-Sánchez,

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.
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References
1. Wang DY, Mao JH, Zhang Y, Gu WZ, Zhao SA et al. Kimura disease: a case report and review of the Chinese literature. Nephron Clin Pr. 2009; 111: c55–61.
2. López Arcas Calleja JM, Iturriaga TM, Romero MP, de María Martínez G, Martín Moro JG et al. Kimura disease: Report of a case in a Caucasian male and revision of the literature. Rev Esp Cir Oral Maxilofac. 2006; 28: 353-8.
3. Abuel-Haija M, Hurford MT. Kimura disease. Arch Pathol Lab Med. 2007; 131(4): 650-651.
4. Sun QF, Xu DZ, Pan SH, Ding JG, Xue ZQ et al. Kimura disease: review of the literatura. Intern Med J. 2008; 38: 668-672.
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Pablo-Martín E, Corvo-Félix L, Rodríguez González M, Jiménez Pérez A, Figuero-Pérez L, Pérez García M, et all. Finding of celiac disease during the study of Kimura disease in a Caucasian male. 10853/2024


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Publication history

Received: 09/10/2024

Accepted: 21/10/2024

Online First: 12/11/2024

Published: 14/01/2026

Article Online First time: 34 days

Article editing time: 462 days


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