Satria Budi, Chen WenChieh, Soebono Hardyanto, Radiono Sunardi, Danarti Retno
Department of Dermatology and Venereology, Faculty of Medicine, Public Health and Nursing, Universitas Gadjah Mada/Dr. Sardjito Hospital, Yogyakarta, Indonesia.
Department of Dermatology and Allergy, Technische Universität München, Munich, Germany.
Case Rep Dermatol. 2019 Sep 10;11(3):240-248. doi: 10.1159/000502509. eCollection 2019 Sep-Dec.
Acrodermatitis enteropathica (AcE) is a rare, autosomal recessive inherited disorder caused by mutation of the gene coding for zinc transport protein (ZIP 4). The disease appears during childhood especially in breastfeeding or post-breastfeeding infant. Eczema herpeticum refers to a disseminated skin infection of herpes simplex virus that usually leads to vesicular eruptions commonly seen on a background of atopic dermatitis (AD). We describe an 11-year-old boy with periorificial erosions in periorbital, perinasal, perioral, perineal, and gluteal areas, accompanied with itchy vesicles, some covered with hemorrhagic crusts. A clinical diagnosis of AcE and eczema herpeticum with AD was supported by typical lesions and acute and chronic eczematous changes found mainly in the flexural aspects of extremities, which is diagnostic of AD. Laboratory findings showed anti HSV1 IgG (23.43) and high levels of IgE (478.9 IU/L). There was no multinucleated giant cell in the Tzanck test. Skin histology was compatible with AcE. Direct immunofluorescent examination showed no deposits of IgG, IgM, IgA, or complement. Complete resolution occurred within 2 weeks of acyclovir and oral zinc supplementation.
肠病性肢端皮炎（AcE）是一种罕见的常染色体隐性遗传性疾病，由编码锌转运蛋白（ZIP 4）的基因突变引起。该疾病在儿童期出现，尤其在母乳喂养期或母乳喂养后的婴儿中。疱疹样湿疹是指单纯疱疹病毒的播散性皮肤感染，通常导致在特应性皮炎（AD）背景下常见的水疱性皮疹。我们描述了一名11岁男孩，其眶周、鼻周、口周、会阴和臀部区域出现口周糜烂，伴有瘙痒性水疱，部分覆盖有出血性痂皮。典型病变以及主要在四肢屈侧发现的急慢性湿疹样改变支持了AcE和伴有AD的疱疹样湿疹的临床诊断，后者可诊断为AD。实验室检查结果显示抗HSV1 IgG（23.43）和高水平的IgE（478.9 IU/L）。Tzanck试验未见多核巨细胞。皮肤组织学与AcE相符。直接免疫荧光检查显示无IgG、IgM、IgA或补体沉积。在使用阿昔洛韦和口服锌补充剂治疗2周内完全缓解。
