Yunus Kaygusuz, Ceren Üstün, Ahmet Görkem Er, Zehranur Kiki, İsmail Yaz, Sevda Tüten-Dal, Ayşegül Üner, Deniz Çağdaş, Ahmet Çağkan İnkaya, Murat Akova
{"title":"由伯克霍尔德氏菌和曲霉菌感染引起的伴有嗜血细胞淋巴组织细胞增多症的成人慢性肉芽肿病病例","authors":"Yunus Kaygusuz, Ceren Üstün, Ahmet Görkem Er, Zehranur Kiki, İsmail Yaz, Sevda Tüten-Dal, Ayşegül Üner, Deniz Çağdaş, Ahmet Çağkan İnkaya, Murat Akova","doi":"10.36519/idcm.2024.381","DOIUrl":null,"url":null,"abstract":"<p><p>Chronic granulomatous disease (CGD) is a congenital disorder impairing phagocyte function, causing recurrent, life-threatening infections, and is rarely seen in adulthood. We present a 36-year-old male initially diagnosed with pneumonia. Bronchoalveolar lavage and blood cultures yielded <i>Burkholderia multivorans/cepacia</i> complex, sputum cultures <i>Aspergillus niger</i>. Despite the antimicrobial treatment, his condition deteriorated. His clinical and laboratory findings indicated hemophagocytic lymphohistiocytosis. He responded to steroids. Nitroblue tetrazolium and dihydroergotamine-123 tests confirmed CGD. Whole exome sequencing identified <i>NCF1</i> deletion. He received interferon-gamma, voriconazole, and trimethoprim-sulfamethoxazole. Allogeneic hematopoietic stem cell transplantation was planned. This case report improves understanding of CGD in adults, aiming to enhance diagnostic and therapeutic strategies.</p>","PeriodicalId":519881,"journal":{"name":"Infectious diseases & clinical microbiology","volume":"6 4","pages":"334-338"},"PeriodicalIF":0.0000,"publicationDate":"2024-12-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11687228/pdf/","citationCount":"0","resultStr":"{\"title\":\"An Adult-Onset Chronic Granulomatous Disease Case with Hemophagocytic Lymphohistiocytosis Caused by <i>Burkholderia</i> and <i>Aspergillus</i> Infections.\",\"authors\":\"Yunus Kaygusuz, Ceren Üstün, Ahmet Görkem Er, Zehranur Kiki, İsmail Yaz, Sevda Tüten-Dal, Ayşegül Üner, Deniz Çağdaş, Ahmet Çağkan İnkaya, Murat Akova\",\"doi\":\"10.36519/idcm.2024.381\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Chronic granulomatous disease (CGD) is a congenital disorder impairing phagocyte function, causing recurrent, life-threatening infections, and is rarely seen in adulthood. We present a 36-year-old male initially diagnosed with pneumonia. Bronchoalveolar lavage and blood cultures yielded <i>Burkholderia multivorans/cepacia</i> complex, sputum cultures <i>Aspergillus niger</i>. Despite the antimicrobial treatment, his condition deteriorated. His clinical and laboratory findings indicated hemophagocytic lymphohistiocytosis. He responded to steroids. Nitroblue tetrazolium and dihydroergotamine-123 tests confirmed CGD. Whole exome sequencing identified <i>NCF1</i> deletion. He received interferon-gamma, voriconazole, and trimethoprim-sulfamethoxazole. Allogeneic hematopoietic stem cell transplantation was planned. This case report improves understanding of CGD in adults, aiming to enhance diagnostic and therapeutic strategies.</p>\",\"PeriodicalId\":519881,\"journal\":{\"name\":\"Infectious diseases & clinical microbiology\",\"volume\":\"6 4\",\"pages\":\"334-338\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2024-12-19\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC11687228/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Infectious diseases & clinical microbiology\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.36519/idcm.2024.381\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2024/12/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Infectious diseases & clinical microbiology","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.36519/idcm.2024.381","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2024/12/1 0:00:00","PubModel":"eCollection","JCR":"","JCRName":"","Score":null,"Total":0}
An Adult-Onset Chronic Granulomatous Disease Case with Hemophagocytic Lymphohistiocytosis Caused by Burkholderia and Aspergillus Infections.
Chronic granulomatous disease (CGD) is a congenital disorder impairing phagocyte function, causing recurrent, life-threatening infections, and is rarely seen in adulthood. We present a 36-year-old male initially diagnosed with pneumonia. Bronchoalveolar lavage and blood cultures yielded Burkholderia multivorans/cepacia complex, sputum cultures Aspergillus niger. Despite the antimicrobial treatment, his condition deteriorated. His clinical and laboratory findings indicated hemophagocytic lymphohistiocytosis. He responded to steroids. Nitroblue tetrazolium and dihydroergotamine-123 tests confirmed CGD. Whole exome sequencing identified NCF1 deletion. He received interferon-gamma, voriconazole, and trimethoprim-sulfamethoxazole. Allogeneic hematopoietic stem cell transplantation was planned. This case report improves understanding of CGD in adults, aiming to enhance diagnostic and therapeutic strategies.