Rasul Naima, Delaney Russell
Medical School, Edward Via College of Osteopathic Medicine, Blacksburg, USA.
Pediatrics Department, Lewis Gale Medical Center, Salem, USA.
Cureus. 2025 Apr 15;17(4):e82333. doi: 10.7759/cureus.82333. eCollection 2025 Apr.
Acute hemorrhagic edema of infancy (AHEI) is a rare cutaneous leukocytoclastic small-vessel vasculitis presenting with purpuric skin plaques on the face, ears, and extremities, frequently associated with fever and edema. Although the condition usually self-resolves over one to three weeks, it can be mistaken for more serious conditions, leading to unnecessary procedures and treatments. This case involves a nine-month-old infant who developed bilateral ear erythema and edema two days after a viral illness and was hospitalized for clinical suspicion of relapsing polychondritis. Within a couple of hours, the infant's erythema and edema progressed, with the appearance of small papules scattered across the right half of the face. Over five days, the patient developed purpuric lesions on the cheeks, chin, bilateral extremities, and trunk. During the hospitalization, the infant received empiric antibiotics, steroids, and antihistamines. Based on the acute edema, scattered purpuric lesions in the setting of a recent viral illness, and negative infectious workup, the infant was diagnosed with AHEI and subsequently managed with steroids to reduce the inflammation rapidly and antihistamines to reduce corticosteroid-induced gastric complications. The ear erythema, ear edema, and purpuric lesions showed resolution 10 days after initial presentation at the outpatient pediatric follow-up. However, while the infant was in good health overall at the outpatient dermatology follow-up approximately three weeks after hospitalization, he did have residual macular spots on the cheeks and bilateral lower extremities. The macular spots disappeared, and the infant had no cutaneous lesions two months after the initial presentation. The resolution of symptoms after oral steroids confirmed the clinical diagnosis of AHEI rather than relapsing polychondritis. This case serves to illustrate an unusual presentation of AHEI to aid in the future diagnosis of infantile rashes and to differentiate this benign diagnosis from other, life-threatening conditions. Early recognition of AHEI could prevent unnecessary treatments or procedures in the future.
婴儿急性出血性水肿(AHEI)是一种罕见的皮肤白细胞破碎性小血管血管炎,表现为面部、耳部和四肢出现紫癜性皮肤斑块,常伴有发热和水肿。尽管该病通常在一至三周内自行消退,但可能被误诊为更严重的疾病,从而导致不必要的检查和治疗。本病例涉及一名9个月大的婴儿,在病毒感染疾病两天后出现双侧耳部红斑和水肿,因临床怀疑复发性多软骨炎而住院。在几个小时内,婴儿的红斑和水肿加重,右半侧面部出现散在的小丘疹。在五天内,患者的脸颊、下巴、双侧四肢和躯干出现紫癜性病变。住院期间,婴儿接受了经验性抗生素、类固醇和抗组胺药治疗。基于急性水肿、近期病毒感染疾病背景下的散在紫癜性病变以及阴性感染检查结果,该婴儿被诊断为AHEI,随后使用类固醇迅速减轻炎症,并使用抗组胺药减少皮质类固醇引起的胃部并发症。在门诊儿科随访中,耳部红斑、耳部水肿和紫癜性病变在首次出现后10天消退。然而,在住院约三周后的门诊皮肤科随访中,尽管婴儿总体健康状况良好,但脸颊和双侧下肢仍有残留的黄斑。黄斑在首次出现后两个月消失,婴儿无皮肤病变。口服类固醇后症状消退证实了AHEI的临床诊断,而非复发性多软骨炎。本病例旨在说明AHEI的一种不寻常表现,以帮助未来诊断婴儿皮疹,并将这种良性诊断与其他危及生命的疾病区分开来。早期识别AHEI可预防未来不必要的治疗或检查。