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秋水仙碱治疗人工血管动静脉内瘘上多发坏疽性脓皮病1例报告

Multiple Pyoderma Gangrenosum Overlying AV Fistula Treated With Colchicine: A Case Report.

作者信息

Derstenfeld Alex, Meunier Rosalie-Sélène, Bouchard Josée, Mereniuk Alexandra

机构信息

Département de Dermatologie, Université de Montréal, QC, Canada.

Département de Médecine, Hôpital du Sacré-Coeur de Montréal, QC, Canada.

出版信息

Can J Kidney Health Dis. 2024 Oct 5;11:20543581241284749. doi: 10.1177/20543581241284749. eCollection 2024.

DOI:10.1177/20543581241284749
PMID:39371934
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11456213/
Abstract

RATIONALE

Pyoderma gangrenosum (PG) is a rare neutrophilic dermatosis which gives rise to painful ulcers. Pyoderma gangrenosum can be triggered by trauma, a phenomenon called pathergy. Here, we report the first case of PG arising from pathergy due to needle insertion overlying an arteriovenous fistula (AVF). This case report seeks to inform nephrologists about PG, this yet unreported presentation, and management in the context of hemodialysis.

PRESENTING CONCERNS

A 69-year-old woman presented to dermatology clinic for erythemato-violaceous plaques with central ulceration at the site of needle insertion overlying her AVF. The patient was known for chronic renal insufficiency secondary to C3 glomerulonephritis, for which she received hemodialysis. After an accidental burn which lead to appearance of a painful ulcer, following each needle insertion for hemodialysis, she would develop an erythematous papule that progressed to a painful ulcer with erythematous-violaceous borders.

DIAGNOSIS

Pyoderma gangrenosum was clinically diagnosed and both clinical and paraclinical evaluation did not reveal any secondary cause of PG.

INTERVENTION

Dialysis via AVF was suspended due to the risk of triggering more PG and was temporarily pursued by central venous catheter. The patient was initially treated with prednisone and topical corticosteroids. Furthermore, owing to the high recurrence rate of PG, colchicine was initiated in prevention to avoid resorting to immunosuppressive or long-term corticotherapy.

OUTCOMES

The patient's lesions improved on prednisone, which was then tapered over 1 month. Following prednisone taper and continuing improvement of PG on colchicine and topical corticosteroids alone, the decision was taken to recommence dialysis via AVF after performing a negative pathergy test. Topical corticosteroids were ceased due to the risk of cutaneous atrophy and were replaced by pimecrolimus ointment. The patient has continued dialysis via AVF ever since, without recurrence.

NOVEL FINDING

This is the first case reported of PG arising from pathergy due to needle insertion overlying an AVF. Colchicine may be a safe and effective therapy for long-term treatment of PG in the context of hemodialysis.

摘要

原理

坏疽性脓皮病(PG)是一种罕见的嗜中性皮肤病,可导致疼痛性溃疡。坏疽性脓皮病可由创伤引发,这一现象称为同形反应。在此,我们报告首例因动静脉瘘(AVF)上方针刺引起同形反应而发生的PG病例。本病例报告旨在让肾病学家了解PG、这种尚未报道的表现形式以及血液透析背景下的治疗方法。

就诊情况

一名69岁女性到皮肤科门诊就诊,其AVF上方针刺部位出现中央溃疡的红斑紫斑。该患者因C3肾小球肾炎继发慢性肾功能不全,正在接受血液透析。一次意外烧伤导致出现疼痛性溃疡后,每次血液透析针刺后,她都会出现一个红斑丘疹,进而发展为边界为红斑紫斑的疼痛性溃疡。

诊断

临床诊断为坏疽性脓皮病,临床和辅助检查均未发现PG的任何继发原因。

干预措施

由于有引发更多PG的风险,暂停通过AVF进行透析,改为临时通过中心静脉导管进行透析。患者最初接受泼尼松和外用皮质类固醇治疗。此外,由于PG复发率高,开始使用秋水仙碱预防,以避免采用免疫抑制或长期皮质激素治疗。

结果

患者的皮损在泼尼松治疗下有所改善,随后在1个月内逐渐减量。在泼尼松减量且仅使用秋水仙碱和外用皮质类固醇后PG持续改善后,在同形反应试验呈阴性后决定重新通过AVF进行透析。由于有皮肤萎缩的风险,停用外用皮质类固醇,改用吡美莫司软膏。此后患者一直通过AVF进行透析,未再复发。

新发现

这是首例因AVF上方针刺引起同形反应而发生PG的病例报告。秋水仙碱可能是血液透析背景下长期治疗PG的安全有效疗法。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8d9/11456213/672fab4b4cea/10.1177_20543581241284749-fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8d9/11456213/2ade65a7c503/10.1177_20543581241284749-fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8d9/11456213/672fab4b4cea/10.1177_20543581241284749-fig2.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8d9/11456213/2ade65a7c503/10.1177_20543581241284749-fig1.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8d9/11456213/672fab4b4cea/10.1177_20543581241284749-fig2.jpg

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