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病例报告:在一名无突变的老年患者中,斯佩索利单抗成功治疗Hallopeau连续性肢端皮炎。

Case report: Successful treatment with spesolimab of acrodermatitis continua of Hallopeau in an older patient without mutations.

作者信息

Zheng Jianfeng, Ding Yangfeng, Shi Yuling, Yi Xuemei

机构信息

Department of Dermatology, Shanghai Skin Disease Hospital, Tongji University School of Medicine, Institue of Psoriasis, Tongji University School of Medicine, Shanghai, China.

出版信息

Front Immunol. 2025 Jan 15;15:1440102. doi: 10.3389/fimmu.2024.1440102. eCollection 2024.

Abstract

BACKGROUND

Acrodermatitis continua of Hallopeau (ACH) is a rare, sterile pustular psoriasis variant refractory to many conventional treatments. The eruption typically occurs after local trauma or infection; other etiologies include neural, inflammatory, and genetic causes. Herein we reported a single case of a 64-year-old patient with ACH that was successfully treated with spesolimab for 19 weeks.

CASE SUMMARY

A 64-year-old Chinese male with no personal or known family history of psoriasis had recurrent episodes of redness, swelling, and pustules in the nail bed on seven fingers with progressive degeneration of the nails. The patient was monitored as to the evolution of the disease over half of a year before he referred his case to our attention. A diagnosis of ACH was made, allowing for the administration of local steroids and oral acitretin. However, after 3 months of acitretin treatment, no improvement was observed. In December 2023, this patient came to our inpatient department; his modified nail psoriasis severity index score was 32. Before starting treatment, a comprehensive set of laboratory and instrumental tests were all found to be negative. Moreover, whole-exome sequencing was performed in our patient, and it revealed no rare coding variant in , , or . Therefore, the patient was administrated with a dose of 900 mg spesolimab. After 10 days, the patient showed a significant decrease in discomfort and pain. In order to strengthen the therapeutic effect, he was given the second dose of 900 mg spesolimab after 4 weeks. After 19 weeks of spesolimab treatment, the patient's nail lesions showed complete resolution, and no adverse effects were reported.

CONCLUSION

The case report suggests that spesolimab may offer significant therapeutic benefits for ACH.

摘要

背景

哈洛佩奥连续性肢端皮炎(ACH)是一种罕见的、无菌性脓疱型银屑病变体,对许多传统治疗方法均难治。皮疹通常在局部创伤或感染后出现;其他病因包括神经、炎症和遗传因素。在此,我们报告了一例64岁的ACH患者,使用司库奇尤单抗成功治疗19周。

病例摘要

一名64岁的中国男性,无个人或已知的银屑病家族史,七个手指的甲床反复出现发红、肿胀和脓疱,指甲逐渐退变。在该患者将其病例提交给我们之前,对其病情演变进行了半年多的监测。诊断为ACH,给予局部类固醇和口服阿维A治疗。然而,阿维A治疗3个月后,未见改善。2023年12月,该患者来到我们的住院部;其改良甲银屑病严重程度指数评分为32。在开始治疗前,所有实验室和器械检查结果均为阴性。此外,对该患者进行了全外显子组测序,结果显示在 、 或 中未发现罕见的编码变异。因此,给予该患者900 mg司库奇尤单抗一剂。10天后,患者的不适和疼痛明显减轻。为加强治疗效果,4周后给予第二剂900 mg司库奇尤单抗。司库奇尤单抗治疗19周后,患者的指甲病变完全消退,且未报告不良反应。

结论

该病例报告表明,司库奇尤单抗可能对ACH具有显著的治疗益处。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/60f2/11774742/8826e0cf2503/fimmu-15-1440102-g001.jpg

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