重新评估法国的 Cladophialophora bantiana 褐丝菌病:全国回顾性研究。
Reappraising Cladophialophora bantiana phaeohyphomycosis in France: retrospective nation-based study.
发表日期:2024 Oct 09
作者:
Olivier Lortholary, Dea Garcia-Hermoso, Aude Sturny-Leclère, Karine Sitbon, Céline Nourrisson, Valérie Letscher-Bru, Nicole Desbois-Nogard, Ferouze Bani-Sadr, Frédéric Bastides, Boris Bienvenu, Camille Cordier, Anne Coste, François Danion, Tristan Dégot, David Delarbre, Arnaud Fekkar, Christophe Garcie, Cyril Garrouste, Maud Gits-Muselli, Emilie Guemas, Antoine Huguenin, Frédéric Janvier, Nassim Kamar, Cyril Kervinio, Solène Le Gal, Olivier Lesens, Marie Machouart, Florence Persat, Sandrine Picot, Anahita Rouze, Stéphane Ranque, Yvon Ruch, Matthieu Saada, Sarah Stabler, Alexandre Alanio, Fanny Lanternier, Guillaume Desoubeaux
来源:
Lancet Microbe
摘要:
Cladophialophora bantiana 是毒性最强的暗丝菌之一,通常会引起非血管生成性单个(或有时多个)囊性脑损伤,并导致死亡率高达 70%。 大多数 C bantiana 病例都是在一系列孤立的报告中或在非常小的群体中描述的。这项全国性回顾性研究的目的是分享过去二十年来法国和法国海外领土报告的 C bantiana 褐丝菌病病例数据,以提高对这种疾病的了解。 C bantiana 感染患者通过主动监测计划进行处理由国家真菌病和抗真菌药物参考中心、巴斯德研究所(法国巴黎)和法国侵袭性真菌感染监测网络发起的侵袭性真菌感染调查,涉及法国大陆和法国海外领土的 29 家医院。根据欧洲癌症和真菌病研究与治疗组织修订和更新的共识定义,该研究仅包括已证实和可能的感染病例。 2002 年至 2022 年间,巴斯德研究所采用多相方法对患者进行诊断或确诊,或两者兼而有之。患者被分为两组:有中枢神经系统受累的患者和无中枢神经系统受累的患者。主要结果是存活率。研究期间(2002 年 1 月 1 日至 2022 年 12 月 31 日)共纳入了 23 名巴蒂亚纳侵入性感染患者。中枢神经系统受累组的中位年龄为 56 岁,非中枢神经系统受累组的中位年龄为 65 岁。直到 2021 年,每年的病例数在 0 到 2 例之间变化,其中 2022 年观察到 6 例病例,这是法国自 1900 年以来有记录以来最热的一年。15 例 (65%) 患者观察到中枢神经系统受累,其中 3 例为播散性病例;七名患者 (30%) 皮肤和软组织受累,一名患者出现孤立性肺部感染。在 5 名患者中观察到糖尿病,在 23 名患者中的 14 名 (61%) 中观察到任何免疫抑制因素。仅考虑中枢神经系统受累的患者时,接受运动或大量引流的患者的 9 个月生存率似乎更高(4 名患者中的 3 名 [75%] 与 11 名患者中的 3 名 [27%];p=0·24),并且显着更高接受三重抗真菌疗法(脂质体两性霉素 B 加泊沙康唑和氟胞嘧啶;9 名患者中的 7 名 [78%] 与 6 名患者中的 1 名 [17%];p=0·040)治疗 2 周或以上的患者。两名患者仅接受切除手术治疗(一名患者成功,另一名患者失访)。这项研究表明,C bantiana 感染的临床表现和潜在医疗状况比之前描述的更加多样化。它还强调了有和没有中枢神经系统受累的患者之间死亡率的显着差异。进行手术并进行三联抗真菌治疗后,预后得到改善。这种罕见且具有破坏性的侵袭性真菌感染应由多学科团队进行管理。Santé Publique France。版权所有 © 2024。由 Elsevier Ltd 出版。
Cladophialophora bantiana is one of the most virulent phaeohyphomycetes, typically causes non-angiogenic single (or sometimes multiple) cystic brain lesions, and has resulted in a mortality rate of up to 70%. Most C bantiana cases are described either in a series of isolated reports or in very small cohorts. The aim of this retrospective nation-based study was to share the data on C bantiana phaeohyphomycosis cases reported in France and French overseas territories over the past two decades to improve understanding of this disease.Patients with C bantiana infection were processed through the active surveillance programme of invasive fungal infections launched by the National Reference Center for Mycoses and Antifungals, Institut Pasteur (Paris, France), and the French Surveillance Network of Invasive Fungal Infections, which involved 29 hospitals from mainland France and overseas French territories. Only proven and probable cases of infection, according to the revised and updated consensus definitions from the European Organization for Research and Treatment of Cancer and Mycoses Study Group, were included in the study. Patients were diagnosed or confirmed, or both, using a polyphasic approach at the Institut Pasteur between 2002 and 2022. Patients were separated into two groups: those with CNS involvement and those with no CNS involvement. The primary outcome was the survival rate.A total of 23 patients with a C bantiana invasive infection were included during the study period (Jan 1, 2002, to Dec 31, 2022). The median age was 56 years in the CNS involvement group and 65 years in the non-CNS involvement group. Until 2021, the annual number of cases varied between zero and two, with six cases observed in 2022, the warmest year recorded in France since 1900. CNS involvement was observed in 15 (65%) patients, including three disseminated cases; skin and soft tissue involvement in seven (30%) patients and an isolated lung infection in one case. Diabetes was observed in five patients, and any immunodepression factor was observed in 14 (61%) of 23 patients. When considering only patients with CNS involvement, 9-month survival appeared higher in patients who underwent exeresis or large drainage (three [75%] of four patients vs three [27%] of 11 patients; p=0·24) and significantly higher in those treated for 2 or more weeks with triple antifungal therapy (liposomal amphotericin B plus posaconazole and flucytosine; seven [78%] of nine patients vs one [17%] of six patients; p=0·040). Two patients were treated with excision surgery alone (one patient with success, and the other patient lost to follow-up).This study shows that the clinical presentations and underlying medical conditions of C bantiana infections are more diverse than previously described. It also emphasises a significant difference in mortality rate between those with and without CNS involvement. The prognosis improved when surgery was performed and triple antifungal therapy was administered. Such rare and devastating invasive fungal infections should be managed by a multidisciplinary team.Santé Publique France.Copyright © 2024. Published by Elsevier Ltd.