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免疫功能正常儿童的侵袭性骨破坏性眼眶曲霉菌病:一例报告

Invasive bony destructive orbital aspergillosis in an immunocompetent child: a case report.

作者信息

Rafizadeh Seyed Mohsen, Mousavi Amir, Rajabi Mohammad Taher, Aghajani Amirhossein, Nozarian Zohreh, Zand Amin

机构信息

Department of Oculo-Facial Plastic and Reconstructive Surgery, Farabi Eye Hospital, Tehran University of Medical Sciences, Tehran, Iran.

Department of Pathology, Farabi Eye Hospital, Tehran University of Medical Sciences, Tehran, Iran.

出版信息

J Ophthalmic Inflamm Infect. 2025 Mar 19;15(1):30. doi: 10.1186/s12348-025-00485-7.

DOI:10.1186/s12348-025-00485-7
PMID:40108050
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11923318/
Abstract

PURPOSE

To report a case of invasive sino-orbital aspergillosis, a rare condition in a healthy child. The patient presented with orbital involvement and bone destruction, an exceedingly uncommon occurrence that mimics other invasive inflammatory or neoplastic orbital lesions.

CASE PRESENTATION

A 4-year-old female presented with an ill-defined, irregular, erythematous mass-like lesion measuring 8 × 10 mm on the left upper eyelid. Orbital computed tomography (CT) revealed an infiltrative soft tissue mass with bone erosions and destruction on the medial side of the frontal bone, extending toward the fronto-maxillary suture in the anterior orbit. Except for the left anterior ethmoidal sinus, the other paranasal sinuses were nearly clear. Magnetic resonance imaging (MRI) showed enhancement of the adjacent dura mater near the site of bony erosion and lesion expansion. The lesion was surgically excised, with drainage of mucopurulent discharge. Pathological examination revealed necrotizing granulomatous inflammation and fungal hyphae, with Aspergillus fumigatus growth confirmed by culture. The patient was diagnosed with invasive orbital aspergillosis. She was treated with intravenous and then oral voriconazole, and there was no recurrence of the disease.

CONCLUSIONS

Invasive orbital aspergillosis with bone destruction of the orbital walls can occur in immunocompetent individuals, including children, without any predisposing factors. It can mimic other invasive orbital diseases, leading to delayed diagnosis and treatment, which may result in life-threatening outcomes if intracranial spread occurs. Therefore, timely orbital biopsy of the lesions is crucial.

摘要

目的

报告一例侵袭性鼻窦眼眶曲霉菌病,这在健康儿童中是一种罕见疾病。该患者表现为眼眶受累及骨质破坏,这种极为罕见的情况可模仿其他侵袭性炎症性或肿瘤性眼眶病变。

病例介绍

一名4岁女性,左上眼睑出现一个边界不清、不规则的红斑样肿块,大小为8×10毫米。眼眶计算机断层扫描(CT)显示为浸润性软组织肿块,额骨内侧有骨质侵蚀和破坏,向前眶延伸至额上颌缝。除左侧前筛窦外,其他鼻窦基本清晰。磁共振成像(MRI)显示在骨质侵蚀和病变扩展部位附近的硬脑膜有强化。病变经手术切除,并排出了脓性分泌物。病理检查显示为坏死性肉芽肿性炎症和真菌菌丝,培养证实有烟曲霉菌生长。该患者被诊断为侵袭性眼眶曲霉菌病。她接受了静脉注射然后口服伏立康唑治疗,疾病未复发。

结论

眼眶壁骨质破坏的侵袭性眼眶曲霉菌病可发生在包括儿童在内的免疫功能正常个体中,且无任何诱发因素。它可模仿其他侵袭性眼眶疾病,导致诊断和治疗延迟,如果发生颅内扩散,可能会导致危及生命的后果。因此,对病变进行及时的眼眶活检至关重要。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/424345b5d95d/12348_2025_485_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/105ed551e320/12348_2025_485_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/a89aaad0264c/12348_2025_485_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/3a6663870c93/12348_2025_485_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/33fe56db38c7/12348_2025_485_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/424345b5d95d/12348_2025_485_Fig5_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/105ed551e320/12348_2025_485_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/a89aaad0264c/12348_2025_485_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/3a6663870c93/12348_2025_485_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/33fe56db38c7/12348_2025_485_Fig4_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d45d/11923318/424345b5d95d/12348_2025_485_Fig5_HTML.jpg

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