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Invasive rhino-orbital-cerebral aspergillosis in an immunocompetent patient.
Journal of Medical Mycology ( IF 3.6 ) Pub Date : 2020-05-19 , DOI: 10.1016/j.mycmed.2020.101002
J Leroy 1 , F Vuotto 2 , V Le 1 , M Cornu 1 , N François 1 , L Marceau 3 , C Fichet 4 , S Loridant 1 , B Sendid 1
Affiliation  

Introduction

Rhino-orbital-aspergillosis (ROA) is a rare but serious disease in immunocompetent patients. Diagnosis is often delayed due to the absence of specific clinical symptoms. We describe the case of a patient who presented initially with ROA which spread progressively to the right ethmoid-sphenoid sinuses and then to the brain.

Observation

A 61-year-old patient with a history of well-controlled diabetes presented with a sudden severe decrease in right visual acuity. Cerebral MRI showed the presence of an infiltrate in the right orbital apex extending to the homolateral cavernous sinus without any cerebral involvement. A diagnosis of right orbital myositis was made and corticosteroid therapy was started. His symptoms worsened progressively leading to quasi-blindness. A new MRI showed the development of right sphenoid-ethmoid osteolytic lesions. A fungal aetiology was suspected and tests for fungal biomarkers found a β-(1-3)-D-glucan level of 99 pg/ml but negative galactomannan. An ethmoid biopsy was performed for histological and mycological investigations, including the detection of Aspergillus DNA by qPCR. qPCR was positive and culture resulted in the isolation of multi-sensitive Aspergillus fumigatus. Treatment was initiated with voriconazole. Due to persistence of blindness and the appearance of a lesion extending to the right frontal lobe, surgical excision was performed followed by antifungal treatment for a total duration of 1 year. The patient is currently stable, but has persistence of blindness in the right eye.

Conclusion

Invasive ROA is a rare but serious disease in immunocompetent patients which should be evoked in the differential diagnosis of a tumour or vasculitis. Early diagnosis is essential for optimal management.



中文翻译:

具有免疫功能的患者的侵袭性鼻-眶-脑曲霉病。

介绍

鼻眶曲霉病(ROA)在免疫功能正常的患者中是一种罕见但严重的疾病。由于缺乏特定的临床症状,诊断通常会延迟。我们描述了一个患者,该患者最初表现为ROA,并逐渐扩散到右筛骨蝶窦,然后扩散到大脑。

观察

一位61岁的糖尿病患者,病史控制得很好,其右视力突然急剧下降。脑MRI显示右眼眶尖处有浸润延伸到同侧海绵窦,没有任何脑部受累。诊断为右眼眶肌炎并开始糖皮质激素治疗。他的症状逐渐恶化,导致半盲。一项新的MRI显示了右蝶窦筛骨溶骨性病变的发展。怀疑是真菌病因,对真菌生物标志物的测试发现β-(1-3)-D-葡聚糖水平为99 pg / ml,而半乳甘露聚糖阴性。进行筛筛活检以进行组织学和真菌学研究,包括曲霉菌的检测通过qPCR的DNA。qPCR呈阳性,培养导致分离出多敏感性烟曲霉。用伏立康唑开始治疗。由于失明的持续性和病变延伸到右额叶的出现,进行了外科切除,然后进行了抗真菌治疗,总持续时间为1年。该患者目前稳定,但右眼一直存在失明。

结论

侵入性ROA在免疫功能正常的患者中是一种罕见但严重的疾病,应在肿瘤或血管炎的鉴别诊断中引起。早期诊断对于优化管理至关重要。

更新日期:2020-05-19
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