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产超广谱β-内酰胺酶大肠埃希菌引起的坏死性筋膜炎作为毛细胞白血病的首发表现:一例报告

Necrotizing Fasciitis by Extended-Spectrum Beta-Lactamase-Producing Escherichia coli as the Initial Presentation of Hairy Cell Leukemia: A Case Report.

作者信息

Pérez-Valdez Diego, Sánchez-Rosado Raúl Rodolfo, Ortiz-Pacheco Lucero Janeth, Villarreal-Zavala Rodrigo, Hernández Alvarado Samuel

机构信息

General Surgery, Hospital General Regional No. 1 "Ignacio García Téllez", Instituto Mexicano del Seguro Social (IMSS), Mérida, MEX.

Colon and Rectal Surgery, Hospital General de México, Mexico City, MEX.

出版信息

Cureus. 2025 Jul 23;17(7):e88614. doi: 10.7759/cureus.88614. eCollection 2025 Jul.

DOI:10.7759/cureus.88614
PMID:40861689
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12373413/
Abstract

Necrotizing fasciitis (NF) is a rapidly progressive and life-threatening soft tissue infection. Fournier's gangrene (FG), a subtype involving the perianal and genital regions, is especially severe in immunocompromised individuals. Although rare, hairy cell leukemia (HCL) can initially present with severe infections due to profound immunosuppression. We report the case of a 53-year-old man with hypertension who developed a perianal abscess caused by extended-spectrum beta-lactamase (ESBL)-producing . Despite surgical drainage and carbapenem therapy, the infection progressed. Laboratory studies revealed pancytopenia, and bone marrow biopsy confirmed HCL with 95% infiltration. Despite initiating rituximab, the patient's condition deteriorated. Imaging showed a large presacral abscess, rectal wall perforation, and perirectal emphysema. Surgical management included diverting loop colostomy, extensive debridement, and abdominal lavage. Postoperatively, the patient developed multiorgan failure and died of septic shock despite intensive care. This case underscores the diagnostic and therapeutic challenges of FG in patients with hematologic malignancies. HCL can present with profound immunosuppression, predisposing to severe, monomicrobial NF caused by multidrug-resistant organisms. ESBL-producing further complicates management. Imaging is essential to assess disease extent, and early surgical intervention is critical. Delay in diagnosing the underlying malignancy and the immunocompromised state contributed to a fatal outcome. FG may be the initial manifestation of occult hematologic malignancy such as HCL. Clinicians should maintain a high index of suspicion for immunosuppressive disorders in patients with severe infections and pancytopenia. Multidisciplinary management, including prompt debridement, broad-spectrum antibiotics, and early hematologic evaluation, is vital to improve outcomes.

摘要

坏死性筋膜炎(NF)是一种迅速进展且危及生命的软组织感染。福尼埃坏疽(FG)是一种累及肛周和生殖器区域的亚型,在免疫功能低下的个体中尤为严重。毛细胞白血病(HCL)虽然罕见,但由于严重免疫抑制,最初可表现为严重感染。我们报告一例53岁高血压男性病例,该患者发生了由产超广谱β-内酰胺酶(ESBL)的细菌引起的肛周脓肿。尽管进行了手术引流和碳青霉烯类治疗,但感染仍进展。实验室检查显示全血细胞减少,骨髓活检确诊为HCL,浸润率达95%。尽管开始使用利妥昔单抗治疗,但患者病情仍恶化。影像学检查显示巨大的骶前脓肿、直肠壁穿孔和直肠周围气肿。手术治疗包括转流性袢状结肠造口术、广泛清创和腹腔灌洗。术后,尽管进行了重症监护,患者仍发生多器官功能衰竭并死于感染性休克。该病例强调了血液系统恶性肿瘤患者中FG的诊断和治疗挑战。HCL可表现为严重免疫抑制,易发生由多重耐药菌引起的严重单微生物NF。产ESBL的细菌使治疗更加复杂。影像学检查对于评估疾病范围至关重要,早期手术干预至关重要。潜在恶性肿瘤和免疫功能低下状态的诊断延迟导致了致命结局。FG可能是隐匿性血液系统恶性肿瘤如HCL的初始表现。临床医生对于严重感染和全血细胞减少的患者应保持对免疫抑制性疾病的高度怀疑。多学科管理,包括及时清创、广谱抗生素治疗和早期血液学评估,对于改善预后至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d912/12373413/91e95dca2518/cureus-0017-00000088614-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d912/12373413/fce57759dff9/cureus-0017-00000088614-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d912/12373413/91e95dca2518/cureus-0017-00000088614-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d912/12373413/fce57759dff9/cureus-0017-00000088614-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d912/12373413/91e95dca2518/cureus-0017-00000088614-i02.jpg

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本文引用的文献

1
Fournier's Gangrene: Microbiological Profile and Risk Factors for Mortality: Review of 97 Cases.福尼尔坏疽:微生物学特征及死亡风险因素:97例病例回顾
Infect Dis Clin Microbiol. 2023 Mar 11;5(1):13-22. doi: 10.36519/idcm.2023.177. eCollection 2023 Mar.
2
Fournier's gangrene and fecal diversion. When, in which patients, and what type should I perform?Fournier 坏疽和粪便转流。何时、何种患者、何种类型应该实施?
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Pathogens and their resistance behavior in necrotizing fasciitis.
坏死性筋膜炎中的病原体及其耐药行为。
Clin Hemorheol Microcirc. 2024;86(1-2):169-181. doi: 10.3233/CH-238119.
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Hairy Cell Leukemia: Where Are We in 2023?毛细胞白血病:2023 年我们处于什么位置?
Curr Oncol Rep. 2023 Aug;25(8):833-840. doi: 10.1007/s11912-023-01419-z. Epub 2023 Apr 25.
5
Perianal Infections in Patients With Hematologic Malignancy: The Risk of Fournier's Gangrene Leading to Mortality and Irreversible Organ Damage.血液恶性肿瘤患者的肛周感染:Fournier 坏疽导致死亡率和不可逆转的器官损伤的风险。
Wounds. 2022 Jan;34(1):31-35. doi: 10.25270/wnds/091421.02. Epub 2021 Sep 14.
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Fournier's Gangrene in Patients with Oncohematological Diseases: A Systematic Review of Published Cases.肿瘤血液系统疾病患者的福尼尔坏疽:已发表病例的系统评价
Healthcare (Basel). 2021 Aug 30;9(9):1123. doi: 10.3390/healthcare9091123.
7
Necrotizing fasciitis caused by mono-bacterial gram-negative infection with - the deadliest of them all: A case series and review of the literature.由单一革兰氏阴性菌感染引起的坏死性筋膜炎——其中最致命的一种:病例系列及文献综述
JPRAS Open. 2021 May 14;29:99-105. doi: 10.1016/j.jpra.2021.04.007. eCollection 2021 Sep.
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A retrospective case series of Fournier's gangrene: necrotizing fasciitis in perineum and perianal region.会阴部和肛周 Fournier 坏疽:坏死性筋膜炎的回顾性病例系列研究。
BMC Surg. 2020 Oct 30;20(1):259. doi: 10.1186/s12893-020-00916-3.
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Guideline for diagnosis and management of hairy cell leukaemia (HCL) and hairy cell variant (HCL-V).毛细胞白血病(HCL)和毛细胞变异型白血病(HCL-V)的诊断与管理指南。
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