Reappraising Cladophialophora bantiana phaeohyphomycosis in France: retrospective nation-based study

IF 20.9 1区生物学 Q1 INFECTIOUS DISEASES

Lancet Microbe Pub Date : 2024-11-01 DOI:10.1016/S2666-5247(24)00139-3

Prof Olivier Lortholary MD , Dea Garcia-Hermoso PhD , Aude Sturny-Leclère PhD , Karine Sitbon MD , Céline Nourrisson PharmD , Valérie Letscher-Bru PharmD , Nicole Desbois-Nogard MD , Prof Ferouze Bani-Sadr MD , Frédéric Bastides MD , Prof Boris Bienvenu MD , Camille Cordier PharmD , Anne Coste MD , François Danion MD , Tristan Dégot MD , David Delarbre MD , Arnaud Fekkar PharmD , Christophe Garcie PharmD , Cyril Garrouste MD , Maud Gits-Muselli MD , Emilie Guemas PharmD , Prof Guillaume Desoubeaux MD

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引用次数: 0

Abstract

Background

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.

Methods

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.

Findings

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).

Interpretation

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.

Funding

Santé Publique France.

查看原文本刊更多论文

重新认识法国的班氏褐斑癣菌病：基于国家的回顾性研究。

背景：Cladophialophora bantiana 是毒性最强的噬菌体之一，通常会引起非血管性的单个（有时是多个）脑囊性病变，死亡率高达 70%。大多数 C bantiana 病例都是在一系列孤立的报告或很小的群体中描述的。这项以国家为基础的回顾性研究旨在分享过去二十年中法国和法国海外领地报告的C bantiana噬菌体病病例数据，以增进对该疾病的了解：通过法国巴黎巴斯德研究所国家真菌和抗真菌药物参考资料中心（National Reference Center for Mycoses and Antifungals, Institut Pasteur, Paris, France）和法国侵袭性真菌感染监测网络（French Surveillance Network of Invasive Fungal Infections）发起的侵袭性真菌感染主动监测计划，对感染 C bantiana 真菌的患者进行处理，该计划涉及法国本土和法国海外领地的 29 家医院。根据欧洲癌症研究与治疗组织和真菌病研究小组修订和更新的共识定义，只有确诊和可能感染的病例才被纳入研究范围。2002年至2022年期间，巴斯德研究所采用多相法对患者进行了诊断或确诊，或同时进行了诊断和确诊。患者分为两组：中枢神经系统受累组和中枢神经系统未受累组。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），共有23名班氏菌侵袭性感染患者被纳入研究范围。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。在2021年之前，每年的病例数在0到2例之间变化，2022年出现了6例病例，这是法国自1900年以来有记录的最温暖的一年。15例（65%）患者出现中枢神经系统受累，其中包括3例散发病例；7例（30%）患者出现皮肤和软组织受累，1例出现孤立的肺部感染。5名患者患有糖尿病，23名患者中有14名（61%）存在免疫抑制因素。如果只考虑中枢神经系统受累的患者，接受切除术或大面积引流术的患者的 9 个月存活率更高（4 例患者中的 3 例[75%] vs 11 例患者中的 3 例[27%]；P=0-24），接受三联抗真菌疗法（脂质体两性霉素 B 加上泊沙康唑和氟尿嘧啶；9 例患者中的 7 例[78%] vs 6 例患者中的 1 例[17%]；P=0-040）治疗 2 周或更长时间的患者的存活率显著更高。两名患者仅接受了切除手术治疗（其中一名患者获得成功，另一名患者失去了随访机会）：这项研究表明，班坦氏菌感染的临床表现和潜在病症比以往描述的更为多样。该研究还强调了中枢神经系统受累和未受累患者之间死亡率的显著差异。手术和三联抗真菌疗法可改善预后。这种罕见的破坏性侵袭性真菌感染应由多学科团队共同处理：资金来源：法国公共卫生部。

本文章由计算机程序翻译，如有差异，请以英文原文为准。

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来源期刊

Lancet Microbe Multiple-

CiteScore

27.20

自引率

0.80%

发文量

278

审稿时长

6 weeks

期刊介绍： The Lancet Microbe is a gold open access journal committed to publishing content relevant to clinical microbiologists worldwide, with a focus on studies that advance clinical understanding, challenge the status quo, and advocate change in health policy.