Cerebral phaeohyphomycosis

  • 文章类型: Review
    Fonsecaeamonophora是Fonsecaea的一种,属于Chaetothyriales。它通常与热带和亚热带地区隔绝,引起反应性炎症,皮肤脓肿,和痛苦。由F.monophora引起的脑感染很少见,但通常是致命的。早期诊断这种疾病很困难,和适当的抗真菌治疗经常被推迟。我们报告了一例53岁的2型糖尿病女性,该女性在2个月前出现头痛,并持续1个月的进行性右侧无力。磁共振成像显示左额叶和call体占位病变。通过手术切除囊性肿块,并且基于BLAST-N搜索中内部转录间隔区域的测序对样品进行鉴定表明,产生最重要比对的序列是F.monophora或类似序列(查询覆盖99%,E值0.0,每标识99.84)。患者接受了3个月的疗程,每天两次伏立康唑,导致完全恢复。
    Fonsecaea monophora is a species of Fonsecaea that belongs to Chaetothyriales. It is usually isolated from tropical and subtropical regions, causing reactive inflammation, skin abscesses, and pain. Cerebral infection caused by F. monophora is rare but often fatal. Diagnosing this disease at an early stage is difficult, and appropriate antifungal therapy is often delayed as a result. We report the case of a 53-year-old woman with type 2 diabetes who presented with a headache 2 months ago and progressive right-sided weakness of 1 month\'s duration. Magnetic resonance imaging revealed a space-occupying lesion in the left frontal lobe and corpus callosum. The cystic mass was removed by surgical intervention, and the identification of the sample based on sequencing of the internal transcribed spaced region in BLAST-N search showed that the sequences producing most significant alignments were F. monophora or similar (query cover 99%, E value 0.0, per ident 99.84). The patient was treated with a 3-month course of twice daily voriconazole, leading to complete recovery.
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  • 文章类型: Case Reports
    目的:扁豆是一种常见的导致脑脓肿的黑色素化真菌。过去,许多病例来自亚洲,尤其是来自印度。迟到了,在亚洲以外的地方,案件有所增加,因此有必要对案件进行审查。
    方法:我们介绍了一例由C.bantiana引起的致命性脑性真菌病,并对2015年至2022年报告的由C.bantiana引起的培养证实的脑脓肿进行了系统评价。
    结果:在发现的39例中,大多数(68%)免疫功能低下。各种临床表现为头痛(53%),偏瘫(34%),视觉障碍(25%),感官改变(18%),失语/构音障碍(12%)和癫痫发作(9%)。在18例(60%)患者中观察到孤立的病变。在发生的顺序中,病变位于额叶(30%),颞叶(27%)和顶叶(20%)区域。共感染5例,如同时检出诺卡氏菌肺炎2例,脑脓肿中的弓形虫DNA,在脑脓肿病例中,共存的新型肺隐球菌感染和共存的念珠菌各1例。84%的病例进行了手术干预。抗真菌治疗包括伏立康唑(80%),两性霉素B脂质体(76%),5-氟胞嘧啶(30%),泊沙康唑(10%),和两性霉素B脱氧胆酸盐(6%)。总死亡率为50%,与亚洲(63.6%)相比,亚洲以外地区的死亡率较低(42%),尽管没有统计学意义。
    结论:C.bantiana脑脓肿是一种全球新兴的感染。下一代测序是即将到来的有希望的诊断测试。早期完全切除病灶并使用有效的抗真菌药物可以改善预后。
    OBJECTIVE: Cladophialophora bantiana is a wonted melanized fungus causing brain abscess. In past many cases were reported from Asia, particularly from India. Of late, there is a rise in cases in places besides Asia and hence a review of the cases is warranted.
    METHODS: We present a case of fatal cerebral phaeohyphomycosis caused by C. bantiana and conduct a systematic review of culture confirmed brain abscess due to C. bantiana reported between 2015 and 2022.
    RESULTS: Of the 39 cases found, majority (68%) were immunocompromised. The various clinical presentations were headache (53%), hemiparesis (34%), visual disturbance (25%), altered sensorium (18%), aphasia/dysarthria (12%) and seizures (9%). Isolated lesion was observed in 18 (60%) patients. In the sequence of occurrence, the lesions were in frontal (30%), temporal (27%) and parietal (20%) region. There were five cases with coinfections such as concurrent detection of Nocardia pneumonia in two cases, toxoplasma DNA in brain abscess, coexisting pulmonary Cryptococcus neoformans infection and coexisting Candida in a case of brain abscess in one case each. Surgical intervention was performed in 84% cases. Antifungal therapy included voriconazole (80%), liposomal amphotericin B (76%), 5-fluorocytosine (30%), posaconazole (10%), and amphotericin B deoxycholate (6%). The overall mortality was 50% with lower mortality (42%) in regions outside Asia compared to Asia (63.6%) though not statistically significant.
    CONCLUSIONS: C. bantiana brain abscess is an emerging infection worldwide. Next generation sequencing is an upcoming promising diagnostic test. Early complete excision of the lesion with effective antifungals may improve the outcome.
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  • 文章类型: English Abstract

    Cladophialophora bantiana is a filamentous fungus, known as a dematiaceous fungus because of the presence of melanin. This fungus is of clinical importance because it is neurotropic and causes cerebral phaeohyphomycosis.
    The available scientific information on the development of cerebral phaeohyphomycosis caused by Cladophialophora bantiana was analysed by selecting articles from the PubMed, Scopus and Google Scholar databases that describe case reports of fungal infection by C. bantiana in adults, taking into account the analysis of the patients\' symptomatology, clinical history and neuroanatomical damage, in addition to considering the mortality of the condition.
    India and United States were the countries with most case reports, with 32 and 11 cases respectively. Moreover, in terms of neuroanatomical lesions, the majority of patients suffered mixed lesions (29%) and frontal lobe lesions (22%). In accordance with the patients\' condition, the pathology has a mortality rate of 62%.
    It is concluded that cerebral phaeohyphomycosis has a high mortality rate, there is no standardised treatment and, in most cases, the fungal infection of the brain is mixed and affects several different parts of it. Furthermore, if not diagnosed and treated in time, it can lead to the patients\' death.
    Infección micótica por Cladophialophora bantiana y desarrollo de feohifomicosis cerebral. Revisión sistemática de 58 informes de caso.
    Introducción. Cladophialophora bantiana es un hongo filamentoso, denominado hongo dematiáceo por la presencia de melanina. Este hongo tiene importancia clínica por ser neurotrópico y causar feohifomicosis cerebral. Material y métodos. Se analizó la información científica disponible sobre el desarrollo de feohifomicosis cerebral provocada por Cladophialophora bantiana, seleccionando artículos de las bases de PubMed, Scopus y Google Scholar, que describen informes de caso sobre infección micótica de C. bantiana en adultos, considerando el análisis de la sintomatología, el historial clínico y los daños neuroanatómicos de los pacientes, además de considerar la mortalidad de la patología. Resultados. La India y Estados Unidos fueron los países con más informes de caso, 32 y 11 casos, respectivamente. Asimismo, en cuanto a las lesiones neuroanatómicas, en su mayoría, los pacientes sufrieron lesiones mixtas (29%) y del lóbulo frontal (22%). De acuerdo con el estado de los pacientes, la patología tiene una mortalidad del 62%. Conclusiones. Se concluye que la feohifomicosis cerebral tiene una alta mortalidad, no existe un tratamiento estandarizado y, en la mayoría de los casos, la infección fúngica del cerebro es mixta y afecta a varias partes del cerebro; además, si no se diagnostica y trata a tiempo, puede ocasionar la muerte de los pacientes.
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  • 文章类型: Journal Article
    UNASSIGNED: Rhinocladiella mackenziei is a neurotropic fungus, which can cause devastating intracerebral infections with up to 100% fatality rate. It is difficult to isolate this fungus in laboratory as it grows slowly and requires diagnostic skills.
    UNASSIGNED: A 42-year-old Pakistani man presented with headache, facial numbness, progressive upper limb weakness, and dysarthria. Magnetic resonance imaging of the brain showed a space-occupying lesion in the basal ganglia region. The patient underwent supratentorial craniotomy for biopsy and excision. Histopathology of the specimen revealed granulomatous inflammation with abscess formation. Periodic acid- Schiff special stains highlighted the presence of numerous septate fungal hyphae. The results revealed the growth of dematiaceous fungi, which were morphologically classified as R. mackenziei. The patient is currently stable and is being on amphotericin and posaconazole, along with neurorehabilitation therapy.
    UNASSIGNED: Rhinocladiella mackenziei brain abscess is a devastating infection with significant mortality. This condition should be suspected in patients with brain abscess from high endemic areas.
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  • 文章类型: Case Reports
    We present a case of brain abscess in an immunocompetent child due to the dematiaceous fungus Microascus cinereus, an organism commonly found in soil and stored grains. The etiologic agent was demonstrated by direct microscopy and culture. The patient responded well to surgical excision of abscess along with a course of amphotericin B and voriconazole. Microascus species have emerged as significant invasive pathogens especially in the immunocompromised patients. To the best of our knowledge, this is the first reported case of brain abscess caused by M. cinereus in an immunocompetent individual with no underlying risk factors.
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  • 文章类型: Case Reports
    Exophiala dermatitidis, a dematiaceous fungus typically found in decaying organic matter worldwide, is a rare cause of fungal infections. Cerebral phaeohyphomycosis is a sporadic but often fatal infection of the brain caused by E. dermatitidis. However, due to limited reports, little is known about its specific predisposing factors, clinical manifestation, and optimal treatment modality. Here, we report a clinical presentation and management of cerebral phaeohyphomycosis in a Chinese patient. An otherwise healthy, young male who was diagnosed with neck fungal lymphadenitis caused by E. dermatitidis 7 months prior and was treated with itraconazole, presented later with progressive intracranial hypertension and persistent coma. Culture of the neck lymphoid tissue produced growth of a black yeast-like fungus, which was identified as E. dermatitidis by sequencing of the ribosomal DNA internal transcribed spacer (ITS) domains. Accordingly, a cerebral biopsy was performed, and the pathological report showed mycelia and fungal granulomas. We also sequenced CARD9 in the patient and found him to be homozygous for loss-of-function mutation; his parents were heterozygous for the same mutation. This is a first case report of cerebral phaeohyphomycosis caused by E. dermatitidis in a CARD9-deficient Chinese patient. He eventually succumbed to brain herniation and severe lung infection with a poor response to therapy. Thus, previously healthy patients with unexplained invasive E. dermatitidis infection, at any age, should be tested for inherited CARD9 deficiency.
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  • 文章类型: Case Reports
    The term phaeohyphomycosis (PHM) means dark-pigmented fungal hyphae. Cerebral PHM (CPHM) with onychomycosis is extremely rare; very few have been reported so far. The authors report a case of CPHM with onychomycosis in a 37-year-old male from a rural background in Haryana, India, with involvement of the left frontal lobe. The mass was resected and biopsy was sent for histopathological examination. He was given antifungal drugs in the postoperative period. The patient responded very well to the treatment, and there were no signs of recurrence at the 6-month follow-up visit. The clinical features, imaging and histopathological investigations, and management of this rare entity are discussed, and the available literature is also reviewed.
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  • 文章类型: Case Reports
    Cerebral phaeohyphomycosis is frequently a fatal disease caused by truly neurotropic dematiaceous fungi. Although rare, this infection occurs especially among immunocompetent patients, and the clinical symptoms are often misdiagnosed as a cerebral tumour or bacterial brain abscess. The appropriate diagnosis and therapy of cerebral infections by melanized fungi are very challenging if they are caused by mysterious fungi with unknown ecological niche. We reported the second case of cerebral phaeohyphomycosis due to Rhinocladiella mackenziei in Iran and the first culture-confirmed case. In this report, the differential diagnosis and histopathological findings are discussed and a review of the literature is provided.
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  • 文章类型: Journal Article
    Cladophialophora bantiana is a phaeoid fungus that only rarely has been isolated from sources other than the human brain. It has a particular tropism for the central nervous system (CNS). We have integrated and updated large-scale data related to several aspects of C. Bantiana and reviewed all the available reports on its cerebral infections, focusing on their geographical distribution, infection routes, immune status of infected individuals, type and location of infections, clinical manifestations and treatment and outcome, briefly looking over the spectrum of other disease entities associated with C. bantiana, that is, extra-cerebral and animal infections and on the environmental sources of this fungus. Among the agents of phaeohyphomycosis, a term used to describe an infection caused by a dark pigmented fungus, C. bantiana has some significant specific features. A total of 120 case reports were identified with a significantly higher percentage of healthy subjects than immune-debilitated patients (58.3% vs. 41.7%). Infections due to C. bantiana occur worldwide. The main clinical manifestations are brain abscess (97.5%), coinfection of brain tissue and meninges (14.2%) and meningitis alone (2.5%). Among immunocompetent patients, cerebral infection occurred in the absence of pulmonary lesions. The mortality rate is 65.0% regardless of the patient\'s immune status. The therapeutic options used include surgery or antifungals alone, and the combination of both, in most cases the fatal outcome being rapid after admission. Since the fungus is a true pathogen, laboratory workers should be made aware that BioSafety Level-3 precautions might be necessary.
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  • 文章类型: Case Reports
    The Fonsecaea species, which are the leading causes of chromoblastomycosis, are not considered neurotropic fungal agents. Fonsecaea pedrosoi is the primary species in the genus and is usually isolated from chromoblastomycosis cases. However, the recently distinguished species F. monophora has been reported in a few cerebral phaeohyphomycosis cases. Here, a case of cerebral phaeohyphomycosis caused by Fonsecaea monophora is presented in a 71-year-old female subject with chronic diabetes mellitus and hypertension. The identification of F. monophora was made through mycological and molecular analysis, and an isolate was differentiated from the closely related F. pedrosoi by sequence data on key bases on the ribosomal internal transcribed spacer region. The case was successfully treated with surgical and medical approaches, and the patient has remained healthy and stable after a ten-month follow up. Given the increasing incidence of this type of infection of the central nervous system (CNS), this case provides further support for the consideration that F. monophora might represent a neurotropic agent.
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