副球孢子菌病与黏膜皮肤利什曼病共感染：一例病例报告及系统综述

文献信息

中文摘要

背景： 系统性真菌病和被忽视的热带病，如副球孢子菌病（PCM）和利什曼病，在拉丁美洲流行地区仍是重大公共卫生问题。尽管这些感染在包括哥伦比亚在内的多个流行国家共享重叠的生态位和危险因素，但其共感染极为罕见且鲜有报道。
方法： 我们报告了首例有文献记载的PCM与黏膜皮肤利什曼病共感染病例，患者为来自哥伦比亚热带流行地区的一名老年男性农业工人。患者表现为上消化道出血、慢性呼吸道症状、长期存在的鼻部和口腔黏膜皮肤病变以及严重营养不良。我们还按照PRISMA指南进行了系统文献综述，以识别截至2026年4月所有先前报道的PCM-利什曼病共感染病例。该综述方案已在开放科学框架（Open Science Framework）注册（https://doi.org/10.17605/OSF.IO/9T5GM）。
结果： 组织病理学检查确立了肺部PCM，额外的鼻部和口腔病变活检证实为黏膜皮肤利什曼病伴肉芽肿性PCM累及。系统综述识别出381条记录，其中仅有两例先前发表的病例符合条件；这三例病例与索引病例一起进行了分析。所有患者均为来自流行地区的成年男性，具有相关环境暴露史，且无证据表明存在公认的免疫抑制性疾病或接受免疫抑制治疗。临床表现高度异质，从局限性黏膜皮肤受累到伴肺部和喉部受累的播散性疾病不等。值得注意的是，哥伦比亚病例通过纳入上消化道出血和严重营养不良扩展了已知的临床谱。在所有病例中，诊断均依赖组织病理学确认，采用两性霉素B继以伊曲康唑治疗获得了良好的临床结局。
结论： PCM-利什曼病共感染是一种罕见但具有临床意义的疾病实体，尤其在流行地区构成重要的诊断挑战。本病例凸显了重叠临床表现所带来的诊断挑战，并强调了对来自两种感染可能共存地区的患者保持高度怀疑并进行全面诊断评估的重要性。

英文摘要

BACKGROUND: Systemic mycoses and neglected tropical diseases, such as paracoccidioidomycosis (PCM) and leishmaniasis, remain major public health concerns in endemic regions of Latin America. Their coinfection is exceptionally rare and scarcely reported although these infections share overlapping ecological niches and risk factors across multiple endemic countries, including Colombia.
METHODS: We report the first documented case of coinfection by PCM and mucocutaneous leishmaniasis in an elderly male agricultural worker from a tropical endemic region in Colombia. The patient presented with upper gastrointestinal bleeding, chronic respiratory symptoms, long-standing nasal and oral mucocutaneous lesions, and severe malnutrition. A systematic literature review was also conducted following PRISMA guidelines to identify all previously reported cases of PCM-leishmaniasis coinfection up to April 2026. The review protocol was registered in Open Science Framework (https://doi.org/10.17605/OSF.IO/9T5GM).
RESULTS: Histopathological examination established pulmonary PCM, and additional nasal and oral lesions biopsies confirmed mucocutaneous leishmaniasis with granulomatous PCM involvement. The systematic review identified 381 records, from which only two previously published cases were eligible; three cases were analyzed together with the index case. All patients were adult males from endemic areas with relevant environmental exposure and no evidence of a recognized immunosuppressive disorder or immunosuppressive therapy. The clinical manifestations were highly heterogeneous, ranging from localized mucocutaneous involvement to disseminated disease with pulmonary and laryngeal compromise. Notably, the Colombian case expanded the known clinical spectrum by including upper gastrointestinal bleeding and severe malnutrition. In all cases, the diagnosis relied on histopathological confirmation, and treatment with amphotericin B followed by itraconazole resulted in favorable clinical outcomes.
CONCLUSION: PCM-leishmaniasis coinfection is a rare but clinically significant entity that poses important diagnostic challenges, particularly in endemic settings. This case highlights the diagnostic challenges posed by overlapping clinical presentations and underscores the importance of maintaining a high index of suspicion and performing comprehensive diagnostic evaluations in patients from regions where both infections may coexist.