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一名糖尿病控制不佳患者发生肺毛霉菌病及支气管食管瘘的罕见病例。

A rare case of pulmonary mucormycosis and broncho-esophageal fistula in a patient with poorly controlled diabetes.

作者信息

Poursadegh Farid, Shazdeh Ahmadi Safieh, Oskouyan Zahra, Alvandi Fard Mohammad Mahdi, Rezaeetalab Fariba, Mozdorian Mahnaz, Basiri Reza

机构信息

Lung Diseases Research Center Mashhad University of Medical Sciences Mashhad Iran.

Resident of cardiology at Mashhad University of Medical Sciences Mashhad Iran.

出版信息

Clin Case Rep. 2024 Jun 28;12(7):e9093. doi: 10.1002/ccr3.9093. eCollection 2024 Jul.

Abstract

KEY CLINICAL MESSAGE

In patients with poorly controlled diabetes, early recognition of rare fungal infections like pulmonary mucormycosis, especially when presenting with unusual complications such as broncho-esophageal fistula, is critical. Prompt intervention with antifungal therapy and consideration for surgical debridement significantly impact outcomes. Multidisciplinary management is paramount for such complex cases.

ABSTRACT

Mucormycosis is a rare fungal infection caused by the Mucorales. This infection is mostly observed among those with poorly controlled diabetes or immunodeficiency. The most common presentation of the infection among those with poorly controlled diabetes is rhino-orbit-cerebral involvement. In this case report, we provide the history and outcome of a rare case of pulmonary mucormycosis in a patient with poorly controlled diabetes who was simultaneously diagnosed with broncho-esophageal fistula. Our patient was a 32-year-old male with a history of poorly controlled diabetes. Over the months, he had complained of productive coughs and dyspnea, which had lately been joined by dysphagia. He also claimed to have lost considerable weight (10 kg) during the previous 3 months. Barium swallow showed an abnormal flow of contrast between the bronchus and esophagus, suggesting a broncho-esophageal fistula. Computed tomography of the thorax revealed a broncho-esophageal fistula between the left main bronchus (LMB) and esophagus. He had a bronchoscopy the next day, which revealed necrosis and a broncho-esophageal fistula in the LMB. A bronchial biopsy showed typical hyphae with necrotic tissue, indicating mucormycosis. The patient's antimycotic medication (liposomal amphotericin) was started and a prompt surgery consult was ordered. The patient, however, passed away from massive hemoptysis. We described a rare case of pulmonary mucormycosis with broncho-esophageal fistula in a patient with poorly controlled diabetes. The rarity of this combination highlights the associated diagnostic and treatment hurdles. Early detection, antifungal medication, as soon as possible surgical debridement of involved tissues, and a multidisciplinary approach could improve patient outcomes.

摘要

关键临床信息

在糖尿病控制不佳的患者中,早期识别罕见的真菌感染,如肺毛霉菌病,尤其是出现支气管食管瘘等异常并发症时,至关重要。及时进行抗真菌治疗并考虑手术清创对治疗结果有显著影响。对于此类复杂病例,多学科管理至关重要。

摘要

毛霉菌病是一种由毛霉目真菌引起的罕见真菌感染。这种感染多见于糖尿病控制不佳或免疫功能低下的患者。糖尿病控制不佳的患者中,该感染最常见的表现是鼻-眶-脑受累。在本病例报告中,我们介绍了一名糖尿病控制不佳且同时被诊断为支气管食管瘘的患者发生罕见肺毛霉菌病的病史及治疗结果。我们的患者是一名32岁男性,有糖尿病控制不佳的病史。数月来,他一直主诉有咳痰和呼吸困难,最近又出现了吞咽困难。他还称在过去3个月内体重减轻了相当多(10千克)。吞钡检查显示支气管和食管之间有造影剂异常流动,提示存在支气管食管瘘。胸部计算机断层扫描显示左主支气管(LMB)和食管之间存在支气管食管瘘。第二天他接受了支气管镜检查,结果显示LMB有坏死和支气管食管瘘。支气管活检显示典型的菌丝和坏死组织,提示为毛霉菌病。开始给患者使用抗真菌药物(脂质体两性霉素),并立即安排了手术会诊。然而,患者因大量咯血死亡。我们描述了一例糖尿病控制不佳的患者发生伴有支气管食管瘘的罕见肺毛霉菌病病例。这种情况的罕见性凸显了相关的诊断和治疗障碍。早期检测、抗真菌药物治疗、尽快对受累组织进行手术清创以及多学科方法可能会改善患者的治疗结果。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/7404/11213688/f95d6bef9e45/CCR3-12-e9093-g001.jpg

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