An Unusual Presentation of Pulmonary Mucormycosis as a “Mass-like” Consolidation in an Infant with Chronic Granulomatous: A Case Report Study

Q3 Medicine
Narges Lashkarbolouk, Mahdi Mazandarani, Ali Ahani Azari, Lobat Shahkar
{"title":"An Unusual Presentation of Pulmonary Mucormycosis as a “Mass-like” Consolidation in an Infant with Chronic Granulomatous: A Case Report Study","authors":"Narges Lashkarbolouk, Mahdi Mazandarani, Ali Ahani Azari, Lobat Shahkar","doi":"10.5812/compreped-136054","DOIUrl":null,"url":null,"abstract":"Introduction: Chronic granulomatous disease (CGD) is a primary hereditary immunodeficiency condition caused by a defect in the nicotinamide adenine dinucleotide phosphate (NADP) oxidase complex. Recurrent infections and inflammatory complications with a narrow bacterial and fungal spectrum characterize CGD. Acute inflammatory and infectious complications are vexing and fatal for them. Therefore, managing CGD with prophylactic antibiotics and antifungal agents is essential. Case Presentation: We present a 23-month-old infant with a chief complaint of recent lymphadenopathy diagnosed with CGD. Two months later, she was hospitalized with uncontrolled fever and lung involvement while receiving prophylactic treatment with trimethoprim/sulfamethoxazole. A mass-like consolidation was seen in her radiography evaluation. Based on the biopsy and bronchoalveolar lavage (BAL) pathology report, pulmonary mucormycosis was confirmed. Two weeks after starting the standard treatment regimen with amphotericin B, the patient’s symptoms improved. She was discharged in good general condition with oral voriconazole and trimethoprim/sulfamethoxazole and referred to do hematopoietic stem cell transplantation (HSCT). Conclusions: Mucormycosis in CGD is not a common infection and usually infects patients with impaired cell-mediated immunity, hematological malignancies, steroid treatment, and diabetes. Patients with CGD are vulnerable when treated with a high dose of immunosuppressant medications. In our case report, the patient was infected with mucormycosis despite not receiving immunosuppressant medications and having no history of hematologic malignancies or diabetes. Our patient also had a mass-like consolidation in her radiological result, a rare feature of pulmonary mucormycosis involvement in CGD patients.","PeriodicalId":37929,"journal":{"name":"Journal of Comprehensive Pediatrics","volume":null,"pages":null},"PeriodicalIF":0.0000,"publicationDate":"2023-05-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Comprehensive Pediatrics","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.5812/compreped-136054","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"Medicine","Score":null,"Total":0}
引用次数: 0

Abstract

Introduction: Chronic granulomatous disease (CGD) is a primary hereditary immunodeficiency condition caused by a defect in the nicotinamide adenine dinucleotide phosphate (NADP) oxidase complex. Recurrent infections and inflammatory complications with a narrow bacterial and fungal spectrum characterize CGD. Acute inflammatory and infectious complications are vexing and fatal for them. Therefore, managing CGD with prophylactic antibiotics and antifungal agents is essential. Case Presentation: We present a 23-month-old infant with a chief complaint of recent lymphadenopathy diagnosed with CGD. Two months later, she was hospitalized with uncontrolled fever and lung involvement while receiving prophylactic treatment with trimethoprim/sulfamethoxazole. A mass-like consolidation was seen in her radiography evaluation. Based on the biopsy and bronchoalveolar lavage (BAL) pathology report, pulmonary mucormycosis was confirmed. Two weeks after starting the standard treatment regimen with amphotericin B, the patient’s symptoms improved. She was discharged in good general condition with oral voriconazole and trimethoprim/sulfamethoxazole and referred to do hematopoietic stem cell transplantation (HSCT). Conclusions: Mucormycosis in CGD is not a common infection and usually infects patients with impaired cell-mediated immunity, hematological malignancies, steroid treatment, and diabetes. Patients with CGD are vulnerable when treated with a high dose of immunosuppressant medications. In our case report, the patient was infected with mucormycosis despite not receiving immunosuppressant medications and having no history of hematologic malignancies or diabetes. Our patient also had a mass-like consolidation in her radiological result, a rare feature of pulmonary mucormycosis involvement in CGD patients.
一例慢性肉芽肿性婴儿肺毛霉菌病“肿块样”合并症的异常表现:病例报告研究
慢性肉芽肿病(CGD)是一种由烟酰胺腺嘌呤二核苷酸磷酸(NADP)氧化酶复合物缺陷引起的原发性遗传性免疫缺陷疾病。复发性感染和炎症并发症与狭窄的细菌和真菌谱是CGD的特征。急性炎症和感染并发症对他们来说是令人烦恼和致命的。因此,用预防性抗生素和抗真菌药物治疗CGD是必不可少的。病例介绍:我们提出了一个23个月大的婴儿,最近的主要主诉淋巴结病诊断为CGD。两个月后,她因发热失控和肺部受累住院,同时接受甲氧苄啶/磺胺甲恶唑预防性治疗。x线检查显示肿块样实变。根据活检和支气管肺泡灌洗(BAL)病理报告,确认肺毛霉菌病。在开始使用两性霉素B的标准治疗方案两周后,患者的症状有所改善。出院时情况良好,口服伏立康唑和甲氧苄氨苄/磺胺甲恶唑,拟行造血干细胞移植。结论:CGD中的毛霉病不是一种常见的感染,通常感染细胞介导免疫受损、血液系统恶性肿瘤、类固醇治疗和糖尿病患者。当使用高剂量的免疫抑制药物治疗时,CGD患者是脆弱的。在我们的病例报告中,患者感染了毛霉菌病,尽管没有接受免疫抑制药物治疗,也没有血液恶性肿瘤或糖尿病史。我们的患者在放射学结果中也有肿块样实变,这是CGD患者肺部毛霉菌病累及的罕见特征。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
求助全文
约1分钟内获得全文 求助全文
来源期刊
Journal of Comprehensive Pediatrics
Journal of Comprehensive Pediatrics Medicine-Pediatrics, Perinatology and Child Health
CiteScore
0.90
自引率
0.00%
发文量
28
期刊介绍: Journal of Comprehensive Pediatrics is the official publication of Iranian Society of Pediatrics (ISP) and a peer-reviewed medical journal which is published quarterly. It is informative for all practicing pediatrics including general medical profession.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:481959085
Book学术官方微信