在一名白种男性的木村病研究中发现乳糜泻。

IF 2.7 4区 医学 Q2 GASTROENTEROLOGY & HEPATOLOGY Revista Espanola De Enfermedades Digestivas Pub Date : 2024-11-12 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
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引用次数: 0

摘要

本病例是一名 27 岁的男性,既往无任何疾病。2023 年 5 月,他因右侧颌下淋巴结肿大 2 厘米、病程长达两个月而被转诊至耳鼻喉科。他没有报告发烧或盗汗,也没有发现口腔或生殖器溃疡。在进行的分析中,他出现了嗜酸性粒细胞增多,左腿和耳轮部位出现斑丘疹性红斑,最初使用局部抗生素和皮质类固醇激素进行治疗。软组织超声检查显示,患者右侧颈后淋巴结有两个肿大,分别位于 I 和 II 层。鉴于怀疑是原发性血液肿瘤,患者接受了PET-CT扫描以完成研究,结果显示双侧颈后淋巴结高代谢。2023 年 6 月,该患者接受了程序性颈椎切开术,完全切除了 Ib 区的右侧主要淋巴结,组织学结果显示为反应性淋巴结炎,伴有强烈的嗜酸性粒细胞增多、血管增生和纤维化。这些结果表明,木村病是第一种可能。由于零星发生的窒息与特定食物无关,外周血检测中 IgE 水平持续升高和嗜酸性粒细胞增多,以及皮肤改变,研究结束时进行了胃镜检查,以排除嗜酸性粒细胞食管炎的存在,结果显示存在侵蚀性十二指肠炎、胃镜检查显示患者患有糜烂性十二指肠炎,绒毛变短,固有层淋巴细胞增多,这些结果与乳糜泻一致,随后,抗内粘膜抗体和转谷氨酰胺酶抗体阳性以及 HLA-DQ8 同源阳性在基因研究中得到证实。患者开始接受皮质类固醇和无麸质饮食的系统治疗,目前仍在继续。
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Finding of celiac disease during the study of Kimura disease in a Caucasian male.

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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来源期刊
CiteScore
2.00
自引率
25.00%
发文量
400
审稿时长
6-12 weeks
期刊介绍: La Revista Española de Enfermedades Digestivas, Órgano Oficial de la Sociedad Española de Patología Digestiva (SEPD), Sociedad Española de Endoscopia Digestiva (SEED) y Asociación Española de Ecografía Digestiva (AEED), publica artículos originales, editoriales, revisiones, casos clínicos, cartas al director, imágenes en patología digestiva, y otros artículos especiales sobre todos los aspectos relativos a las enfermedades digestivas.
期刊最新文献
A rare early gastric mixed neuroendocrine-non-neuroendocrine neoplasms misdignosed to early gastric cancer. Acute acalculous cholecystitis secondary to hepatitis A infection. Brief description of Mendelian randomized studies. Eosinophilic gastroenteritis: a rare cause of ileitis. Intestinal graft-versus-host disease.
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