Nonepisodic angioedema with eosinophilia following a rose-thorn prick injury

Kimitoshi Matsuo , Hidenori Takahashi , Hiroki Nagamatsu , Ryutaro Hirose , Yuka Yamada , Hiroaki Takei , Naoya Toba , Mio Toyama-Kousaka , Shinichiro Ota , Miwa Morikawa , Masaharu Shinkai
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Abstract

Background

Nonepisodic angioedema with eosinophilia (NEAE) is a condition marked by angioedema and significant eosinophilia and often linked with atopic dermatitis. It predominantly affects young Asian women and occurs more frequently in the autumn and winter. Despite over 100 reported cases, its etiology and pathogenesis remain unclear.

Case presentation

A 23-year-old Japanese female florist presented with acute arm swelling following rose-thorn pricks to her hands and fingers in spring. One week later, she developed progressive symmetrical non-pitting edema in her lower legs and a 3 kg weight gain without any rash. She had a history of oral allergy syndrome to apples and pears for which allergen-specific IgE were previously detected. Blood tests showed significant eosinophilia (14,930 cells/μL) and elevated thymus and activation-regulated chemokine (TARC) levels (12,864 pg/mL). Thyroid disease, autoimmune disorders, and hematologic malignancies were ruled out. Normal cardiac markers and a whole-body computed tomography excluded visceral organ involvement. She was diagnosed with NEAE and treated with oral prednisolone, which resolved the edema within 10 days. Prednisolone was tapered gradually on an outpatient basis without recurrence.

Conclusion

A review of the literature indicates that NEAE triggered by subcutaneous antigen exposure may not follow the typical age or seasonal patterns. Direct subcutaneous antigen exposure, including rose-thorn pricks, can trigger NEAE. Clinicians should consider NEAE in atypical presentations and thoroughly investigate preceding episodes.

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玫瑰刺伤后伴有嗜酸性粒细胞增多的非阵发性血管性水肿
背景嗜酸性粒细胞增多性非阵发性血管性水肿(NEAE)是一种以血管性水肿和明显的嗜酸性粒细胞增多为特征的疾病,通常与特应性皮炎有关。它主要影响年轻的亚洲女性,多发于秋冬季节。病例介绍 一位 23 岁的日本女性花店店员在春季被玫瑰刺伤手部和手指后出现急性手臂肿胀。一周后,她的小腿出现进行性对称性非点状水肿,体重增加了 3 公斤,但未出现任何皮疹。她曾有过对苹果和梨的口腔过敏综合征病史,之前曾检测出过敏原特异性 IgE。血液化验显示她有明显的嗜酸性粒细胞增多(14,930 个细胞/μL),胸腺和活化调节趋化因子(TARC)水平升高(12,864 pg/mL)。排除了甲状腺疾病、自身免疫性疾病和血液系统恶性肿瘤的可能性。心脏指标正常,全身计算机断层扫描排除了内脏器官受累。她被诊断为NEAE,并接受了口服泼尼松龙治疗,水肿在10天内得到缓解。结论 文献综述表明,皮下抗原暴露引发的 NEAE 可能并不遵循典型的年龄或季节模式。包括玫瑰刺在内的直接皮下抗原暴露可诱发 NEAE。临床医生应考虑非典型表现的 NEAE,并对之前的发作进行彻底调查。
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