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造血细胞移植后慢性肉芽肿病患儿发生侵袭性真菌感染并累及脑部

Invasive Fungal Infection by with Cerebral Involvement in a Pediatric Patient Affected by Chronic Granulomatous Disease After Hematopoietic Cell Transplant.

作者信息

Garonzi Chiara, Chinello Matteo, Caddeo Giulia, Bonetti Elisa, Esposto Maria Pia, Pezzella Vincenza, Vitale Virginia, Zaccaron Ada, Sorrentino Annarita, Gibellini Davide, Cesaro Simone

机构信息

Pediatric Hematology Oncology, Department of Mother and Child, Azienda Ospedaliera Universitaria Integrata Verona, 37126 Verona, Italy.

Microbiology Unit, Azienda Ospedaliera Universitaria Integrata Verona, 37126 Verona, Italy.

出版信息

J Fungi (Basel). 2025 Apr 1;11(4):270. doi: 10.3390/jof11040270.

DOI:10.3390/jof11040270
PMID:40278093
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC12028247/
Abstract

A 5-year-old boy affected by chronic granulomatous disease (CGD) underwent two allogeneic hematopoietic cell transplants (HCT) from the same unrelated donor. The first HCT was complicated by prolonged fever and primary graft failure. While fully aplastic, the patient developed a disseminated infection by involving the knee and parasternal skin (day +34 and +40 post-HCT). The patient was treated with voriconazole and granulocyte transfusions followed by a second HCT 80 days after the first HCT. At day +105, the patient developed fever, headache, and altered level of consciousness associated with multiple bilateral cerebral abscesses at magnetic resonance imaging. The serum B-D-glucan test was positive. Micafungin was added to voriconazole. Despite an initial clinical improvement, the patient developed hydrocephalus. was cultured from cerebrospinal fluid. Liposomal amphotericin B, instead of micafungin, was combined with voriconazole as salvage therapy. Unfortunately, the patient developed uncal herniation and died at day +193 from HCT. This case shows that the prognosis of scedosporiosis remains poor despite adequate antifungal treatment. Noteworthy, the B-D-Glucan test is confirmed useful as a non-invasive marker for early diagnosis and may help the differential diagnosis of mycoses.

摘要

一名患有慢性肉芽肿病(CGD)的5岁男孩接受了来自同一无关供体的两次异基因造血细胞移植(HCT)。第一次HCT出现了持续发热和原发性移植失败等并发症。在完全再生障碍期间,患者在HCT后第34天和第40天出现了累及膝盖和胸骨旁皮肤的播散性感染。患者接受了伏立康唑和粒细胞输注治疗,在第一次HCT后80天进行了第二次HCT。在第105天,患者出现发热、头痛和意识水平改变,磁共振成像显示有多个双侧脑脓肿。血清β-D-葡聚糖检测呈阳性。在伏立康唑基础上加用了米卡芬净。尽管最初临床症状有所改善,但患者出现了脑积水。脑脊液培养出了[具体病原体未给出]。作为挽救治疗,用脂质体两性霉素B代替米卡芬净与伏立康唑联合使用。不幸的是,患者出现了枕骨大孔疝,并在HCT后第193天死亡。该病例表明,尽管进行了充分的抗真菌治疗,赛多孢子菌病的预后仍然很差。值得注意的是,β-D-葡聚糖检测被证实是一种有用的早期诊断非侵入性标志物,可能有助于真菌病的鉴别诊断。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/cba2bf975466/jof-11-00270-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/ba0fa1628422/jof-11-00270-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/b151e98d0ed8/jof-11-00270-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/617f51982e28/jof-11-00270-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/cba2bf975466/jof-11-00270-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/ba0fa1628422/jof-11-00270-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/b151e98d0ed8/jof-11-00270-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/617f51982e28/jof-11-00270-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d53b/12028247/cba2bf975466/jof-11-00270-g004.jpg

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本文引用的文献

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Transpl Infect Dis. 2025 Jan-Feb;27(1):e14397. doi: 10.1111/tid.14397. Epub 2024 Nov 11.
2
Update on therapeutic approaches for invasive fungal infections in adults.成人侵袭性真菌感染治疗方法的最新进展。
Ther Adv Infect Dis. 2024 Jan 20;11:20499361231224980. doi: 10.1177/20499361231224980. eCollection 2024 Jan-Dec.
3
HCT alleviates disease burden in CGD.
羟基脲减轻慢性肉芽肿病的疾病负担。
Blood. 2023 Dec 14;142(24):2043-2045. doi: 10.1182/blood.2023021538.
4
Genotype, oxidase status, and preceding infection or autoinflammation do not affect allogeneic HCT outcomes for CGD.基因型、氧化酶状态以及先前的感染或自身炎症反应并不影响慢性肉芽肿病(CGD)的异基因造血细胞移植(HCT)结果。
Blood. 2023 Dec 14;142(24):2105-2118. doi: 10.1182/blood.2022019586.
5
Performance of the beta-glucan test for the diagnosis of invasive fusariosis and scedosporiosis: a meta-analysis.贝塔葡聚糖检测对侵袭性镰刀菌病和枝顶孢霉病诊断性能的荟萃分析。
Med Mycol. 2023 Jul 6;61(7). doi: 10.1093/mmy/myad061.
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J Biomed Sci. 2023 Jun 19;30(1):42. doi: 10.1186/s12929-023-00926-2.
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