将阿维A酸作为育龄妇女鱼鳞病的一种治疗方式:病例系列和文献综述。

IF 3 3区 医学 Q2 DERMATOLOGY
Dermatology Pub Date : 2024-01-01 Epub Date: 2023-09-04 DOI:10.1159/000533934
Julia Clabbers, Noor van van Oosten, Marieke Bolling, Maaike Vreeburg, Michel van Geel, Peter Steijlen, Antoni Gostynski
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引用次数: 0

摘要

背景:阿维A酸是一种人工合成的维生素A衍生物,是目前研究最多、应用最广泛的治疗鱼鳞病的口服维甲酸类药物。它的主要缺点是停药后三年内需要采取避孕措施。育龄妇女需要另一种替代品。对于另一种维甲酸类药物阿维A酸,停药1个月后怀孕被认为是安全的:本研究旨在提供证据,证明阿维A酸可替代阿西曲汀治疗育龄妇女的鱼鳞病。我们通过一个病例系列分享了我们的经验,并对目前的文献进行了综述:九名育龄妇女(19-31 岁,中位数 21 岁)患有不同亚型的鱼鳞病(常染色体隐性先天性鱼鳞病、(表皮)表皮溶解性鱼鳞病、变异性红皮病和表皮溶解性表皮痣,表皮溶解性鱼鳞病的一种镶嵌形式),她们在 2-28 个月期间接受了 30 毫克阿维A酸的治疗。鱼鳞病的严重程度通过鱼鳞病面积严重性指数(IASI)和研究者全面评估(IGA)来衡量。使用 "阿维A酸"、"遗传性皮肤病 "和 "鱼鳞病 "等关键词在Pubmed上进行了文献检索:结果:IGA、IASI-红斑、IASI-鳞屑和 IASI-总分的平均值显著降低。7 名患者仍在接受治疗,1 名患者因怀孕而停止治疗,1 名患者因经济原因停止治疗。观察到的副作用有可逆性头痛(6 例)、骨化性湿疹(1 例)、暂时 "不舒服"(1 例)和脚部更容易起泡(1 例)。通过文献检索,我们找到了六篇有关阿维A酸治疗鱼鳞病和鱼鳞病综合征的病例报告和系列病例,共有29名患者接受了治疗。绝大多数文章(21/29)都报道了皮肤症状明显改善甚至完全缓解的情况。然而,这些结果缺乏经过验证的结果指标来支持。除良性颅内高压(1 例)和自身免疫性甲状腺功能减退(1 例)外,副作用(16 例)相对较轻:我们的研究结果表明,阿维A酸能有效缓解育龄妇女的鱼鳞病症状,是阿西曲汀的合适替代品。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Alitretinoin as a Treatment Modality for Ichthyosis in Women of Childbearing Age: A Case Series and Review of the Literature.

Background: Acitretin, a synthetic vitamin A derivative, is the most studied and widely used oral retinoid for ichthyoses. Its major disadvantage is the need for contraceptive measures during 3 years after discontinuation. An alternative is needed for women of childbearing age. With alitretinoin, another retinoid, pregnancy is considered safe 1 month after discontinuation.

Objectives: The aim of this study was to provide evidence for alitretinoin as an alternative for acitretin for ichthyosis in women of childbearing age. Our experience is shared in a case series combined with an overview of the current literature.

Methods: Nine women of childbearing age (19-31 years, median 21) with different subtypes of ichthyosis (autosomal recessive congenital ichthyosis, (superficial) epidermolytic ichthyosis, erythrokeratoderma variabilis, and epidermolytic epidermal nevi, a mosaic form of epidermolytic ichthyosis) were included and treated with 30 mg alitretinoin during 2-28 months. Severity was measured by Ichthyosis Area Severity Index (IASI) and Investigator Global Assessment (IGA). A literature search in Pubmed using the Mesh terms "alitretinoin," "skin diseases, genetic" and "ichthyosis" was performed.

Results: Significant reduction in the mean scores of IGA, IASI-erythema, IASI-scaling, and IASI-total was seen. Seven patients are still being treated, 1 patient stopped to become pregnant, 1 patient discontinued due to financial reasons. Observed side effects were reversible headache (n = 6), asteatotic eczema (n = 1), "not feeling well" temporarily (n = 1), and easier blistering of the feet (n = 1). The literature search resulted in six case reports and case series about alitretinoin in ichthyosis and ichthyosis syndromes with in total 29 patients. The vast majority of articles (21/29) reported significant improvement or even complete remission of skin symptoms. However, validated outcome measures to support these results were lacking. Side effects (n = 16) were relatively mild, except for benign intracranial hypertension (n = 1) and autoimmune hypothyroidism (n = 1).

Conclusion: Our study shows, with validated outcome measures, that alitretinoin is effective to mitigate the symptoms of ichthyosis in women of childbearing age and a suitable alternative to acitretin.

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来源期刊
Dermatology
Dermatology 医学-皮肤病学
CiteScore
6.40
自引率
2.90%
发文量
71
审稿时长
1 months
期刊介绍: Published since 1893, ''Dermatology'' provides a worldwide survey of clinical and investigative dermatology. Original papers report clinical and laboratory findings. In order to inform readers of the implications of recent research, editorials and reviews prepared by invited, internationally recognized scientists are regularly featured. In addition to original papers, the journal publishes rapid communications, short communications, and letters to ''Dermatology''. ''Dermatology'' answers the complete information needs of practitioners concerned with progress in research related to skin, clinical dermatology and therapy. The journal enjoys a high scientific reputation with a continually increasing impact factor and an equally high circulation.
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