Zelieskova Maria, Banovcin Peter, Kozar Marek, Kozarova Andrea, Nudzajova Zuzana, Jesenak Milos
Department of Paediatrics, Centre for Primary Immunodeficiencies, Jessenius Faculty of Medicine of Comenius University in Bratislava, University Hospital in Martin, Martin, Slovakia.
Department of Neonatology, Jessenius Faculty of Medicine of Comenius University in Bratislava, University Hospital in Martin, Martin, Slovakia.
Pediatr Dermatol. 2020 Nov;37(6):1202-1204. doi: 10.1111/pde.14318. Epub 2020 Aug 7.
We report a 2-year-old patient with Netherton syndrome presenting with generalized exfoliative erythroderma, ichthyosiform dermatitis, trichorrhexis invaginata, hypernatremic dehydration, failure to thrive, and recurrent respiratory infections. Molecular analysis of SPINK5 identified a novel mutation (c.1530CA). Our case report also verifies and supports the safety and efficacy of subcutaneous immunoglobulin substitution in chronic generalized skin disorders associated with primary immunodeficiencies such as Netherton syndrome.
我们报告了一名患有Netherton综合征的2岁患者,其表现为全身性剥脱性红皮病、鱼鳞病样皮炎、套叠性脆发症、高钠血症性脱水、生长发育迟缓以及反复呼吸道感染。对SPINK5的分子分析鉴定出一种新的突变(c.1530C>A)。我们的病例报告还验证并支持了皮下免疫球蛋白替代疗法在与原发性免疫缺陷相关的慢性全身性皮肤疾病(如Netherton综合征)中的安全性和有效性。