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遗传性皮肤疾病伴异色性皮肤症。

Inherited skin disorders presenting with poikiloderma.

机构信息

Department of Dermatology and Venereology, Faculty of Medicine, Public Health, and Nursing, Universitas Gadjah Mada, Yogyakarta, Indonesia.

Skin Research Institute of Singapore, Agency for Science, Technology and Research (A*STAR), Singapore, Singapore.

出版信息

Int J Dermatol. 2021 Nov;60(11):1343-1353. doi: 10.1111/ijd.15498. Epub 2021 Mar 19.

DOI:10.1111/ijd.15498
PMID:33739439
Abstract

Poikiloderma is a skin condition that combines atrophy, telangiectasia, and macular pigment changes (hypo- as well as hyperpigmentation). It is often mistaken for mottled pigmentation by general practitioners or nondermatology specialists. Poikiloderma can be a key presenting symptom of Rothmund-Thomson syndrome (RTS), dyskeratosis congenita (DC), hereditary sclerosing poikiloderma (HSP), hereditary fibrosing poikiloderma with tendon contractures, myopathy, and pulmonary fibrosis (POIKTMP), xeroderma pigmentosum (XP), Bloom syndrome (BS), Kindler syndrome (KS), and Clericuzio-type poikiloderma with neutropenia (PN). In these conditions, poikiloderma starts early in life, usually before the second or third year. They may also be associated with photosensitivity and other significant multi-organ manifestation developed later in life. Poikiloderma could indicate the presence of a genetic disorder with potentially serious consequences. Poikiloderma almost always precedes more severe manifestations of these genodermatoses. Prompt diagnosis at the time of presentation could help to prevent complications and mitigate the course of the disease. This review discusses these to help the practicing clinician manage patients presenting with the symptom. To further facilitate early recognition, this paper also proposes a simple diagnostic algorithm.

摘要

斑驳病是一种皮肤疾病,其特征为萎缩、毛细血管扩张和黄斑色素改变(色素减退和色素沉着)。它常被全科医生或非皮肤科专家误诊为斑驳色素沉着。斑驳病可能是 Rothmund-Thomson 综合征 (RTS)、先天性角化不良 (DC)、遗传性硬化性斑驳病 (HSP)、遗传性纤维性斑驳病伴肌腱挛缩、肌病和肺纤维化 (POIKTMP)、着色性干皮病 (XP)、Bloom 综合征 (BS)、Kindler 综合征 (KS) 和 Clericuzio 型伴中性粒细胞减少的斑驳病的主要表现症状。在这些疾病中,斑驳病通常在生命的第二年或第三年前开始出现,而且可能与光敏感性和其他重要的多器官表现有关,这些表现会在以后的生活中出现。斑驳病可能表明存在具有潜在严重后果的遗传疾病。斑驳病几乎总是先于这些遗传性皮肤病更严重的表现。在出现症状时及时诊断可以帮助预防并发症并减轻疾病的进程。本综述讨论了这些疾病,以帮助临床医生管理出现这种症状的患者。为了进一步促进早期识别,本文还提出了一个简单的诊断算法。

相似文献

1
Inherited skin disorders presenting with poikiloderma.遗传性皮肤疾病伴异色性皮肤症。
Int J Dermatol. 2021 Nov;60(11):1343-1353. doi: 10.1111/ijd.15498. Epub 2021 Mar 19.
2
Mutation in FAM111B Causes Hereditary Fibrosing Poikiloderma with Tendon Contracture, Myopathy, and Pulmonary Fibrosis.FAM111B基因突变导致遗传性纤维化性皮肤异色症伴肌腱挛缩、肌病和肺纤维化。
Acta Derm Venereol. 2019 Jun 1;99(7):695-696. doi: 10.2340/00015555-3186.
3
Kindler syndrome.金德勒综合征
Indian J Dermatol Venereol Leprol. 2005 Sep-Oct;71(5):348-50. doi: 10.4103/0378-6323.16788.
4
Rothmund-Thomson syndrome.Rothmund-Thomson 综合征。
Orphanet J Rare Dis. 2010 Jan 29;5:2. doi: 10.1186/1750-1172-5-2.
5
A case of hereditary fibrosing poikiloderma with tendon contractures, myopathy and pulmonary fibrosis (POIKTMP) with the emphasis on cutaneous histopathological findings.1例伴有肌腱挛缩、肌病和肺纤维化的遗传性纤维性皮肤异色症(POIKTMP),重点关注皮肤组织病理学表现。
J Eur Acad Dermatol Venereol. 2018 Dec;32(12):e443-e445. doi: 10.1111/jdv.14968. Epub 2018 May 1.
6
CUGC for hereditary fibrosing poikiloderma with tendon contractures, myopathy, and pulmonary fibrosis (POIKTMP).遗传性纤维性苔藓样皮肤异色症伴肌腱挛缩、肌病和肺纤维化(POIKTMP)的CUGC
Eur J Hum Genet. 2016 May;24(5). doi: 10.1038/ejhg.2015.205. Epub 2015 Oct 7.
7
Expanding the clinical spectrum of hereditary fibrosing poikiloderma with tendon contractures, myopathy and pulmonary fibrosis due to FAM111B mutations.因FAM111B基因突变导致的伴有肌腱挛缩、肌病和肺纤维化的遗传性纤维性皮肤异色症临床谱的扩展。
Orphanet J Rare Dis. 2015 Oct 15;10:135. doi: 10.1186/s13023-015-0352-4.
8
Poikiloderma with neutropenia, Clericuzio type, in a family from Morocco.来自摩洛哥一个家族的Cléricuzio型伴有中性粒细胞减少的皮肤异色症。
Am J Med Genet A. 2008 Nov 1;146A(21):2762-9. doi: 10.1002/ajmg.a.32524.
9
Dermatological manifestations of hereditary fibrosing poikiloderma with tendon contractures, myopathy and pulmonary fibrosis (POIKTMP): a case series of 28 patients.伴有肌腱挛缩、肌病和肺纤维化的遗传性纤维化性皮肤异色症(POIKTMP)的皮肤表现:28例病例系列
Br J Dermatol. 2019 Oct;181(4):862-864. doi: 10.1111/bjd.17996. Epub 2019 Apr 10.
10
Mutations in C16orf57 and normal-length telomeres unify a subset of patients with dyskeratosis congenita, poikiloderma with neutropenia and Rothmund-Thomson syndrome.C16orf57 基因突变和正常长度端粒将一组先天性角化不良、伴有中性粒细胞减少的斑驳皮肤和 Rothmund-Thomson 综合征患者统一起来。
Hum Mol Genet. 2010 Nov 15;19(22):4453-61. doi: 10.1093/hmg/ddq371. Epub 2010 Sep 3.

引用本文的文献

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Ubiquitin-proteasome system dysregulation in FAM111B-related poikiloderma and phenotypic spectrum expansion: new case reports and long-term follow-up.泛素-蛋白酶体系统失调在FAM111B相关的皮肤异色病及表型谱扩展中的作用:新病例报告及长期随访
EBioMedicine. 2025 Aug 20;119:105864. doi: 10.1016/j.ebiom.2025.105864.
2
Case Report: Diverse phenotypes of congenital poikiloderma associated with mutations in codon 628: A case report and literature review.病例报告:与628密码子突变相关的先天性皮肤异色症的多样表型:一例病例报告及文献综述
Front Genet. 2022 Aug 25;13:926451. doi: 10.3389/fgene.2022.926451. eCollection 2022.
3
Mutations in the ribosome biogenesis factor gene LTV1 are linked to LIPHAK syndrome, a novel poikiloderma-like disorder.
核糖体生物发生因子基因 LTV1 中的突变与 LIPHAK 综合征有关,这是一种新的类鱼鳞病样疾病。
Hum Mol Genet. 2022 Jun 22;31(12):1970-1978. doi: 10.1093/hmg/ddab368.