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妊娠期合并克罗恩病患者并发 Fournier 坏疽。

Fournier's Gangrene During Pregnancy in a Patient with Crohn's Disease.

机构信息

Colorectal Surgery Division, Department of Gastroenterology, Hospital das Clinicas HCFMUSP, Faculty of Medicine, University of São Paulo, São Paulo, SP, Brazil.

出版信息

Am J Case Rep. 2022 Mar 24;23:e934942. doi: 10.12659/AJCR.934942.

DOI:10.12659/AJCR.934942
PMID:35321999
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8958861/
Abstract

BACKGROUND Fournier's gangrene (FG) is a rapidly progressive necrotizing infection of the perineum. Risk factors include male sex and immunosuppression. Inflammatory bowel disease and pregnancy may alter immune response by complex mechanisms but have rarely been associated with necrotizing infections of the perineum. To the best of our knowledge, only 5 cases of FG in association with IBD have been reported in the literature, and none of them occurred during pregnancy. CASE REPORT We report the case of a young woman with long-standing Crohn's disease in clinical remission with Infliximab monotherapy who developed FG in the third trimester of pregnancy. A cesarean section was undertaken at 35 weeks due to fetal distress, followed by debridement, diverting stoma, and vacuum-assisted therapy. The perineal defect was closed following 4 debridements and vacuum-therapy exchanges with a unilateral medial thigh advancement flap, and a draining seton was placed in the suprasphincteric fistula. The patient was discharged after 28 days and her recovery was unremarkable. The neonate also recovered well. CONCLUSIONS The treatment of FG is multidisciplinary and includes early debridement and intestinal diversion. Perianal pain should not be disregarded, as it may be the initial symptom of severe perianal sepsis in the immunosuppressed. To the best of our knowledge, this is the first case report of FG during pregnancy in a patient with Crohn's disease.

摘要

背景

Fournier 坏疽(FG)是一种会迅速发展的会阴部坏死性感染。危险因素包括男性和免疫抑制。炎症性肠病和妊娠可能通过复杂的机制改变免疫反应,但很少与会阴部的坏死性感染有关。据我们所知,文献中仅报道了 5 例与 IBD 相关的 FG,且均未发生在妊娠期间。

病例报告

我们报告了一例患有长期缓解期克罗恩病的年轻女性,她接受英夫利昔单抗单药治疗,在妊娠晚期发生 FG。由于胎儿窘迫,在 35 周时进行了剖宫产,随后进行清创、转流造口术和真空辅助治疗。通过 4 次清创和真空治疗与单侧大腿内侧推进皮瓣交换,会阴缺损得以闭合,并在括约肌上瘘管中放置引流缝线。患者在 28 天后出院,恢复情况良好。新生儿也恢复良好。

结论

FG 的治疗是多学科的,包括早期清创和肠道转流。不应忽视肛周疼痛,因为它可能是免疫抑制患者严重肛周脓毒症的初始症状。据我们所知,这是首例在克罗恩病患者妊娠期间发生 FG 的病例报告。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/f6405036bb39/amjcaserep-23-e934942-v001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/e872dc52f99b/amjcaserep-23-e934942-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/eb3527bb7251/amjcaserep-23-e934942-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/83ffa289b601/amjcaserep-23-e934942-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/f6405036bb39/amjcaserep-23-e934942-v001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/e872dc52f99b/amjcaserep-23-e934942-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/eb3527bb7251/amjcaserep-23-e934942-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/83ffa289b601/amjcaserep-23-e934942-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f811/8958861/f6405036bb39/amjcaserep-23-e934942-v001.jpg

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本文引用的文献

1
Crohn's disease.克罗恩病。
Nat Rev Dis Primers. 2020 Apr 2;6(1):22. doi: 10.1038/s41572-020-0156-2.
2
Fournier's gangrene in an obese female in third trimester of pregnancy.一名妊娠晚期肥胖女性的福尼尔坏疽。
Saudi Med J. 2018 Apr;39(4):415-418. doi: 10.15537/smj.2018.4.21780.
3
Necrotizing Soft-Tissue Infections.坏死性软组织感染
一名患有溃疡性结肠炎的女性患者的福尼尔坏疽复杂病例的管理。
Tech Coloproctol. 2024 Nov 12;28(1):155. doi: 10.1007/s10151-024-03032-z.
4
Fournier's Gangrene Associated With Untreated Crohn's Disease in a Male Patient: A Case Report and Review of the Literature.男性患者中与未治疗的克罗恩病相关的福尼尔坏疽:一例报告并文献复习
Cureus. 2024 Aug 22;16(8):e67515. doi: 10.7759/cureus.67515. eCollection 2024 Aug.
5
A Comprehensive Literature Review of Fournier's Gangrene in Females.女性福尼尔坏疽的综合文献综述
Cureus. 2023 May 12;15(5):e38953. doi: 10.7759/cureus.38953. eCollection 2023 May.
N Engl J Med. 2017 Dec 7;377(23):2253-2265. doi: 10.1056/NEJMra1600673.
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European Hernia Society guidelines on prevention and treatment of parastomal hernias.欧洲疝学会关于造口旁疝预防与治疗的指南
Hernia. 2018 Feb;22(1):183-198. doi: 10.1007/s10029-017-1697-5. Epub 2017 Nov 13.
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Fournier gangrene in pregnancy.妊娠期福尼埃坏疽
Obstet Gynecol. 2015 Jun;125(6):1342-1344. doi: 10.1097/AOG.0000000000000678.
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Pregnancy and infection.怀孕与感染。
N Engl J Med. 2014 Sep 11;371(11):1077. doi: 10.1056/NEJMc1408436.
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Necrotizing fasciitis complicating pregnancy: a case report and literature review.坏死性筋膜炎合并妊娠:一例病例报告及文献综述
Case Rep Obstet Gynecol. 2014;2014:505410. doi: 10.1155/2014/505410. Epub 2014 Mar 9.
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Drug-specific risk of non-tuberculosis opportunistic infections in patients receiving anti-TNF therapy reported to the 3-year prospective French RATIO registry.报告至为期 3 年的前瞻性法国 RATIO 注册研究的接受抗 TNF 治疗患者的非结核性机会性感染的药物特异性风险。
Ann Rheum Dis. 2011 Apr;70(4):616-23. doi: 10.1136/ard.2010.137422. Epub 2010 Dec 21.
9
Fournier's gangrene complicating ulcerative pancolitis.并发于溃疡性全结肠炎的 Fournier 坏疽。
J Crohns Colitis. 2010 Jun;4(2):203-6. doi: 10.1016/j.crohns.2009.11.006. Epub 2009 Dec 21.
10
Fournier gangrene: a review of 41 patients and strategies for reconstruction.福尼尔坏疽:41例患者回顾及重建策略
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