Matsuo Kimitoshi, Takahashi Hidenori, Nagamatsu Hiroki, Hirose Ryutaro, Yamada Yuka, Takei Hiroaki, Toba Naoya, Toyama-Kousaka Mio, Ota Shinichiro, Morikawa Miwa, Shinkai Masaharu
Department of Respiratory Medicine, Tokyo Shinagawa Hospital, Shinagawa-ku, Tokyo, Japan.
Division of Respiratory Medicine, Department of Internal Medicine, Toho University Ohashi Medical Center, Meguro-ku, Tokyo, Japan.
Int J Cardiol Cardiovasc Risk Prev. 2024 Jun 18;22:200301. doi: 10.1016/j.ijcrp.2024.200301. eCollection 2024 Sep.
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.
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.
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.
非周期性嗜酸性粒细胞增多性血管性水肿(NEAE)是一种以血管性水肿和显著嗜酸性粒细胞增多为特征的疾病,常与特应性皮炎相关。它主要影响年轻亚洲女性,在秋冬季节更常见。尽管已有100多例报道,但该病的病因和发病机制仍不清楚。
一名23岁的日本女性花店店员,春季手部和手指被玫瑰刺刺伤后出现手臂急性肿胀。一周后,她双下肢出现进行性对称性非凹陷性水肿,体重增加3千克,无任何皮疹。她有对苹果和梨的口腔过敏综合征病史,之前检测到过敏原特异性IgE。血液检查显示显著嗜酸性粒细胞增多(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。正常的心脏标志物和全身计算机断层扫描排除了内脏器官受累。她被诊断为NEAE,并接受口服泼尼松龙治疗,10天内水肿消退。泼尼松龙在门诊逐渐减量,未复发。
文献综述表明,由皮下抗原暴露引发的NEAE可能不遵循典型的年龄或季节模式。包括玫瑰刺刺伤在内的直接皮下抗原暴露可引发NEAE。临床医生应考虑非典型表现中的NEAE,并对先前发作进行全面调查。