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肾病综合征患儿播散性曲霉病的挑战性治疗

Challenging Treatment of Disseminated Aspergillosis in a Child With Nephrotic Syndrome.

作者信息

Dizi Işik Aylin, Akkoç Gülşen, Ergenç Zeynep, Yılmaz Seyhan, Aslan Tuncay Sevgi, Parlak Burcu, Canizci Erdemli Pınar, Büyüktaş Aytaş Didem, Abacı Çapar M Çağla, Sönmez Özcan, Dağçınar Adnan, Öcal Demir Sevliya, Kepenekli Eda

机构信息

From the Division of Pediatric Infectious Diseases, Department of Pediatrics, Marmara University School of Medicine, İstanbul, Türkiye.

Department of Neurosurgery, Marmara University School of Medicine, İstanbul, Türkiye.

出版信息

Pediatr Infect Dis J. 2024 Oct 1;43(10):e363-e365. doi: 10.1097/INF.0000000000004422. Epub 2024 Jun 10.

DOI:10.1097/INF.0000000000004422
PMID:38865571
Abstract

INTRODUCTION

Central nervous system (CNS) aspergillosis is an opportunistic infection with an increasing incidence and a high mortality rate. It is seen in immunocompromised patients as well as in immunocompetent patients. Here, we present disseminated aspergillosis in a child with nephrotic syndrome treated with long-term and aggressive systemic antifungal treatment and intraventricular (IVent) liposomal amphotericin B (L-AmB) as well as surgical excision and drainage due to difficulty in management.

CASE REPORT

A 10-year-old boy with nephrotic syndrome on steroid therapy was admitted with limping and weakness. The cranial magnetic resonance imaging showed multiple intraparenchymal scattered abscesses. The largest one was excised and drained. Abscess culture revealed Aspergillus fumigatus and histopathological examination revealed septate hyphae compatible with Aspergillosis. Intravenous (IV) voriconazole was started, and IV L-AmB was added. The size of lesions and perilesional edema continued to increase, and then IVent L-AmB was added. With IVent and systemic antifungal treatment, regression of the lesions was observed. He was followed up with oral voriconazole and weekly IVent L-AmB. After 2 and a half months, he was re-operated because of increased lesion size, number and perilesional edema, and IV voriconazole and other salvage antifungal therapies were started. Since the lesions had decreased and remained stable, IV voriconazole was switched to oral therapy, and he was followed up as an outpatient. Immunodeficiency diseases were excluded by immunological and genetic tests.

CONCLUSION

Management of central nervous system aspergillosis can be challenging despite long-term and aggressive systemic and IVent antifungal treatment as well as surgical excision and drainage.

摘要

引言

中枢神经系统曲霉病是一种机会性感染,发病率不断上升且死亡率很高。免疫功能低下患者以及免疫功能正常患者均可发生。在此,我们报告1例肾病综合征患儿发生播散性曲霉病,因其病情难以控制,接受了长期积极的全身抗真菌治疗、脑室内(IVent)注射脂质体两性霉素B(L-AmB)以及手术切除和引流。

病例报告

一名接受类固醇治疗的10岁肾病综合征男孩因跛行和乏力入院。头颅磁共振成像显示脑实质内有多个散在脓肿。最大的脓肿被切除并引流。脓肿培养发现烟曲霉,组织病理学检查显示有与曲霉病相符的有隔菌丝。开始静脉注射伏立康唑,并加用静脉注射L-AmB。病变大小和病灶周围水肿持续增加,随后加用脑室内注射L-AmB。通过脑室内和全身抗真菌治疗,观察到病变有所消退。对其进行口服伏立康唑和每周脑室内注射L-AmB的随访。2个半月后,因病变大小、数量增加以及病灶周围水肿加重再次进行手术,开始静脉注射伏立康唑和其他挽救性抗真菌治疗。由于病变已缩小并保持稳定,静脉注射伏立康唑改为口服治疗,作为门诊患者进行随访。通过免疫学和基因检测排除了免疫缺陷疾病。

结论

尽管进行了长期积极的全身和脑室内抗真菌治疗以及手术切除和引流,但中枢神经系统曲霉病的治疗仍具有挑战性。

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