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一个新的角蛋白丝聚集素基因致病性变异导致弥漫性炎症性剥脱性皮肤综合征伴显著嗜酸性粒细胞增多和发内卷曲。

A novel pathogenic variant in the corneodesmosin gene causing generalized inflammatory peeling skin syndrome with marked eosinophilia and trichorrhexis invaginata.

机构信息

Department of Dermatology, Starship Children's Health, Auckland District Health Board, Auckland, New Zealand.

Genetic Health Service New Zealand (Northern Hub), Auckland, New Zealand.

出版信息

Pediatr Dermatol. 2022 Mar;39(2):268-272. doi: 10.1111/pde.14939. Epub 2022 Feb 17.

Abstract

Generalized inflammatory peeling skin syndrome (PSS) is a rare autosomal recessive genodermatosis caused by loss-of-function disease-causing variants of the corneodesmosin gene (CDSN), resulting in excessive shedding of the superficial layers of the epidermis. We describe a case of generalized inflammatory PSS in an infant, presenting at day two of life with ichthyosiform erythroderma and superficial peeling of the skin. Hair microscopy showed trichorrhexis invaginata. Normal amounts of skin LEKT1, a product of SPINK5 on immunohistochemical staining excluded a diagnosis of Netherton syndrome. Genetic analysis revealed a homozygous novel complete CDSN deletion, estimated 4.6 kb in size, supporting the diagnosis of generalized inflammatory PSS.

摘要

全身性炎症性剥脱性皮肤综合征(PSS)是一种罕见的常染色体隐性遗传皮肤病,由角蛋白丝聚集素基因(CDSN)的功能丧失性致病变异引起,导致表皮浅层过度脱落。我们描述了一例婴儿全身性炎症性 PSS 病例,该患儿在出生后第二天出现鱼鳞样红皮病和皮肤浅层剥脱。毛发显微镜检查显示发内凹。免疫组织化学染色显示皮肤 LEKT1 正常,丝氨酸蛋白酶抑制剂 Kazal 型 5(SPINK5)的产物正常,排除了 Netherton 综合征的诊断。基因分析显示一个纯合的新型 CDSN 完全缺失,大小估计为 4.6 kb,支持全身性炎症性 PSS 的诊断。

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