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瘙痒性大疱性表皮松解症:1例伴有新突变及并存苔藓样淀粉样变的病例

Epidermolysis bullosa pruriginosa: a case with a novel mutation and co-existent lichen amyloidosus.

作者信息

Chen Qiping, Lee Joyce Siong-See, Tey Hong Liang

机构信息

National Skin Centre, 1 Mandalay Road, Singapore.

出版信息

Indian J Dermatol Venereol Leprol. 2015 Jan-Feb;81(1):40-2. doi: 10.4103/0378-6323.148565.

DOI:10.4103/0378-6323.148565
PMID:25566895
Abstract

Epidermolysis bullosa pruriginosa is a rare variant of dystrophic epidermolysis bullosa characterized by severely pruritic and cicatricial lesions localized to the extensor extremities. We report a Singaporean Chinese male with epidermolysis bullosa pruriginosa with an underlying novel mutation in the COL7A1 gene. A heterozygous acceptor splice site mutation IVS67-1G>T probably led to in-frame skipping of exon 68 (36-basepairs), resulting in a loss of 12 amino acids. Among his three children, only the youngest son, who had bilateral big toenail thickening, possessed the same mutation. His skin biopsy one decade ago revealed association of focal amyloidosis; a recent skin biopsy showed more established features of lichen amyloidosis. It is debatable whether the cutaneous amyloidosis was a secondary or primary phenomenon. Our report highlights that the diagnosis of epidermolysis bullosa pruriginosa may be obscured when cutaneous amyloidosis is coexistent.

摘要

瘙痒性大疱性表皮松解症是营养不良性大疱性表皮松解症的一种罕见变体,其特征为严重瘙痒且瘢痕性皮损局限于四肢伸侧。我们报告了一名患有瘙痒性大疱性表皮松解症的新加坡华裔男性,其COL7A1基因存在一种潜在的新突变。一个杂合的受体剪接位点突变IVS67 - 1G>T可能导致外显子68(36个碱基对)的框内跳跃,导致12个氨基酸缺失。在他的三个孩子中,只有最小的儿子有双侧大脚趾甲增厚,且具有相同的突变。他十年前的皮肤活检显示有局灶性淀粉样变;最近的皮肤活检显示有更典型的苔藓样淀粉样变特征。皮肤淀粉样变是继发性还是原发性现象仍有争议。我们的报告强调,当皮肤淀粉样变共存时,瘙痒性大疱性表皮松解症的诊断可能会受到影响。

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Epidermolysis bullosa pruriginosa: a case with a novel mutation and co-existent lichen amyloidosus.瘙痒性大疱性表皮松解症:1例伴有新突变及并存苔藓样淀粉样变的病例
Indian J Dermatol Venereol Leprol. 2015 Jan-Feb;81(1):40-2. doi: 10.4103/0378-6323.148565.
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引用本文的文献

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Nail involvement in patients with epidermolysis bullosa: A systematic review.大疱性表皮松解症患者的指甲受累:一项系统综述。
Skin Health Dis. 2022 Nov 10;3(1):e183. doi: 10.1002/ski2.183. eCollection 2023 Feb.