Secukinumab作为年轻人慢性内瑟顿综合征的新疗法。

IF 1 Q3 MEDICINE, GENERAL & INTERNAL
Irma Buchukuri, Giorgi Chalatashvili, Maia Gotua, Kakhaber Robakidze, Nino Khutsishvili, Nino Lortkipanidze
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引用次数: 0

摘要

内瑟顿综合征是一种常染色体隐性遗传病,通常诊断于婴儿期,其特征为鱼鳞病、红皮病、特应性和特征性毛干珠状(内伏毛癣)。Secukinumab是一种抑制白细胞介素-17的单克隆抗体,最近被报道对难治性内瑟顿综合征患者有效。本报告描述了一名患有慢性难治性内瑟顿综合征的20岁男性,他对secukinumab治疗有反应。病例报告一名20岁男性患者,表现为全身瘙痒、红斑和脱屑。患者先前被诊断为特应性皮炎,并接受全身皮质类固醇和omalizumab治疗,但未见改善。后来,病人被转介到我们这里。体格检查显示全身性红皮病和多环和蛇形红斑斑块,边缘有双刃鳞片。头皮毛镜检查发现发干异常,特别是内阴毛漏。血液检查显示IgE水平升高。最初,患者开始使用局部皮质类固醇,这导致病情恶化。由于患者对治疗没有反应,因此决定开始使用secukinumab单药治疗。2次注射后患者病情明显好转,末次注射后疗效维持4-5个月。结论内瑟顿综合征是一种罕见且易误诊的皮肤病遗传性疾病。因此,该疾病的管理策略是可变的,没有共识。在我们的病例中,考虑到疾病的发病机制,我们使用了secukinumab,它已经显示出很好的效果和良好的耐受性。需要进一步的研究来确定这种药物的功效。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Secukinumab as a Novel Treatment for Chronic Netherton Syndrome in a Young Adult.

BACKGROUND Netherton syndrome is an autosomal recessive skin disorder usually diagnosed in infancy and is characterized by ichthyosis, erythroderma, atopy, and characteristic beading of the hair shaft (trichorrhexis invaginata). Secukinumab, a monoclonal antibody that inhibits interleukin-17, has recently been reported to be effective in patients with refractory Netherton syndrome. The present report describes a 20-year-old man with a chronic history of refractory Netherton syndrome who responded to treatment with secukinumab. CASE REPORT A 20-year-old male patient presented with generalized pruritus, erythema, and scaling. The patient was previously diagnosed with atopic dermatitis and had been treated with systemic corticosteroids and omalizumab, without improvement. Later, the patient was referred to us. Physical examination showed generalized erythroderma and polycyclic and serpiginous erythematous plaques with double-edged scales at the margins. Trichoscopy of the scalp revealed a hair shaft abnormality, specifically trichorrhexis invaginata. The blood tests demonstrated elevated IgE levels. Initially, the patient was started on topical corticosteroids, which caused the condition to deteriorate. As the patient did not respond to the treatment, a decision was made to start secukinumab monotherapy. After 2 injections, the patient's condition markedly improved and the effect after the last injection was maintained for 4-5 months. CONCLUSIONS Netherton syndrome is a rare and often misdiagnosed hereditary diseases in dermatology. For this reason, the management strategies of the disease are variable and without consensus. In our case, considering the pathogenesis of the disease, we used secukinumab, which has shown promising results and good tolerability. Further studies are required to establish the efficacy of the drug.

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来源期刊
American Journal of Case Reports
American Journal of Case Reports Medicine-Medicine (all)
CiteScore
1.80
自引率
0.00%
发文量
599
期刊介绍: American Journal of Case Reports is an international, peer-reviewed scientific journal that publishes single and series case reports in all medical fields. American Journal of Case Reports is issued on a continuous basis as a primary electronic journal. Print copies of a single article or a set of articles can be ordered on demand.
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