Aylin Dizi Işik, Gülşen Akkoç, Zeynep Ergenç, Seyhan Yılmaz, Sevgi Aslan Tuncay, Burcu Parlak, Pınar Canizci Erdemli, Didem Büyüktaş Aytaş, M Çağla Abacı Çapar, Özcan Sönmez, Adnan Dağçınar, Sevliya Öcal Demir, Eda Kepenekli
{"title":"肾病综合征患儿播散性曲霉菌病的治疗难题。","authors":"Aylin Dizi Işik, Gülşen Akkoç, Zeynep Ergenç, Seyhan Yılmaz, Sevgi Aslan Tuncay, Burcu Parlak, Pınar Canizci Erdemli, Didem Büyüktaş Aytaş, M Çağla Abacı Çapar, Özcan Sönmez, Adnan Dağçınar, Sevliya Öcal Demir, Eda Kepenekli","doi":"10.1097/INF.0000000000004422","DOIUrl":null,"url":null,"abstract":"<p><strong>Introduction: </strong>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.</p><p><strong>Case report: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":19858,"journal":{"name":"Pediatric Infectious Disease Journal","volume":" ","pages":"e363-e365"},"PeriodicalIF":2.9000,"publicationDate":"2024-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Challenging Treatment of Disseminated Aspergillosis in a Child With Nephrotic Syndrome.\",\"authors\":\"Aylin Dizi Işik, Gülşen Akkoç, Zeynep Ergenç, Seyhan Yılmaz, Sevgi Aslan Tuncay, Burcu Parlak, Pınar Canizci Erdemli, Didem Büyüktaş Aytaş, M Çağla Abacı Çapar, Özcan Sönmez, Adnan Dağçınar, Sevliya Öcal Demir, Eda Kepenekli\",\"doi\":\"10.1097/INF.0000000000004422\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><strong>Introduction: </strong>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.</p><p><strong>Case report: </strong>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.</p><p><strong>Conclusion: </strong>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.</p>\",\"PeriodicalId\":19858,\"journal\":{\"name\":\"Pediatric Infectious Disease Journal\",\"volume\":\" \",\"pages\":\"e363-e365\"},\"PeriodicalIF\":2.9000,\"publicationDate\":\"2024-10-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Pediatric Infectious Disease Journal\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1097/INF.0000000000004422\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/6/10 0:00:00\",\"PubModel\":\"Epub\",\"JCR\":\"Q3\",\"JCRName\":\"IMMUNOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Pediatric Infectious Disease Journal","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1097/INF.0000000000004422","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/6/10 0:00:00","PubModel":"Epub","JCR":"Q3","JCRName":"IMMUNOLOGY","Score":null,"Total":0}
引用次数: 0
摘要
简介中枢神经系统(CNS)曲霉菌病是一种机会性感染,发病率越来越高,死亡率也很高。它既见于免疫力低下的患者,也见于免疫力正常的患者。在此,我们介绍了一名肾病综合征患儿的播散性曲霉菌病,由于治疗困难,该患儿接受了长期、积极的全身抗真菌治疗和静脉注射两性霉素 B 脂质体(L-AmB)以及手术切除和引流治疗:一名患有肾病综合征、正在接受类固醇治疗的 10 岁男孩因跛行和虚弱入院。头颅磁共振成像显示多发性实质内散在脓肿。最大的一个脓肿被切除并引流。脓肿培养发现了烟曲霉菌,组织病理学检查发现了与曲霉菌病相符的隔膜菌丝。开始静脉注射伏立康唑,并加用左旋氨苄。皮损面积和皮损周围水肿继续扩大,随后又增加了静脉注射 L-AmB。通过静脉注射和全身抗真菌治疗,观察到皮损消退。他接受了口服伏立康唑和每周静脉滴注 L-AmB 的随访。两个半月后,由于皮损面积、数量和周围水肿增加,他再次接受了手术,并开始静脉注射伏立康唑和其他挽救性抗真菌治疗。由于病灶缩小并保持稳定,静脉注射伏立康唑改为口服治疗,并对他进行了门诊随访。免疫学和基因检测排除了免疫缺陷疾病:尽管需要长期、积极的全身和静脉抗真菌治疗以及手术切除和引流,但中枢神经系统曲霉菌病的治疗仍具有挑战性。
Challenging Treatment of Disseminated Aspergillosis in a Child With Nephrotic Syndrome.
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.
期刊介绍:
The Pediatric Infectious Disease Journal® (PIDJ) is a complete, up-to-the-minute resource on infectious diseases in children. Through a mix of original studies, informative review articles, and unique case reports, PIDJ delivers the latest insights on combating disease in children — from state-of-the-art diagnostic techniques to the most effective drug therapies and other treatment protocols. It is a resource that can improve patient care and stimulate your personal research.