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中华妇幼临床医学杂志(电子版) ›› 2018, Vol. 14 ›› Issue (04) : 406 -412. doi: 10.3877/cma.j.issn.1673-5250.2018.04.006

所属专题: 经典病例 文献

论著

儿童血液系统恶性疾病合并侵袭性镰刀菌病1例并文献复习
廖雄宇1, 邱坤银1, 吴若豪1, 黄科1, 方建培1, 周敦华1,()   
  1. 1. 510120 广州,中山大学孙逸仙纪念医院儿科
  • 收稿日期:2018-03-12 修回日期:2018-06-15 出版日期:2018-08-01
  • 通信作者: 周敦华

Hematologic malignancies combined with invasive fusariosis: a case report and literatures review

Xiongyu Liao1, Kunyin Qiu1, Ruohao Wu1, Ke Huang1, Jianpei Fang1, Dunhua Zhou1,()   

  1. 1. Department of Pediatrics, Sun Yat-Sen Memorial Hospital, Sun Yat-Sen University, Guangzhou 510120, Guangdong Province, China
  • Received:2018-03-12 Revised:2018-06-15 Published:2018-08-01
  • Corresponding author: Dunhua Zhou
  • About author:
    Corresponding author: Zhou Dunhua, Email:
  • Supported by:
    Science and Technology Developmental Project of Department of Science and Technology of Guangdong Province(2017A020215086)
引用本文:

廖雄宇, 邱坤银, 吴若豪, 黄科, 方建培, 周敦华. 儿童血液系统恶性疾病合并侵袭性镰刀菌病1例并文献复习[J/OL]. 中华妇幼临床医学杂志(电子版), 2018, 14(04): 406-412.

Xiongyu Liao, Kunyin Qiu, Ruohao Wu, Ke Huang, Jianpei Fang, Dunhua Zhou. Hematologic malignancies combined with invasive fusariosis: a case report and literatures review[J/OL]. Chinese Journal of Obstetrics & Gynecology and Pediatrics(Electronic Edition), 2018, 14(04): 406-412.

目的

探讨儿童血液系统恶性疾病合并侵袭性镰刀菌病的临床特点及其治疗方案,并进行文献复习。

方法

选择2016年7月15日,于中山大学孙逸仙纪念医院儿童血液病区住院治疗的1例骨髓增生异常综合征(MDS)合并侵袭性镰刀菌病的13岁女性患儿为研究对象,对其临床特点进行分析。文献复习设定检索策略为:以"恶性""血液系统疾病""侵袭性镰刀菌病""malignant""hematologic disease"及"invasive fuasriosis"为关键词,在PubMed数据库、中国生物医学文献服务系统(SinoMed)、中国知网(CNKI)、万方数据知识服务平台和维普中文科技期刊数据库进行相关文献检索,收集并分析检索到的血液系统恶性疾病合并侵袭性镰刀菌病的相关文献,检索年限设定为2010年1月1日至2016年12月31日。总结儿童血液系统恶性疾病合并侵袭性镰刀菌病的临床特点及其诊治方法。本研究符合2013年修订的《世界医学协会赫尔辛基宣言》的要求。

结果

①根据患儿临床表现及相关实验室检查结果,诊断为MDS。化疗后,其胸部CT检查可见肺部浸润影,随后四肢、躯干出现紫红色硬结伴中央坏死,并且四肢出现多个皮下液性包块。皮肤活组织病理学检查、骨髓培养和包块引流物培养均提示镰刀菌感染,针对镰刀菌感染采取两性霉素B+泊沙康唑抗真菌治疗策略。随访患儿至2017年4月5日,因患儿原发基础疾病无法缓解,家属放弃治疗出院。②共计检索到国内外相关文献7篇,涉及21例血液系统恶性疾病合并侵袭性镰刀菌病患儿,其中男性患儿为12例,女性为9例;主要临床表现为持续性发热并且积极化疗+抗感染治疗无效,预后较差。21例患儿中,死亡为14例,治愈为7例。

结论

血液系统恶性疾病合并镰刀菌病患儿应早诊断,并且治疗血液系统恶性疾病的同时,给予有效和足量、足疗程的抗真菌药物治疗。两性霉素B联合泊沙康唑可作为治疗侵袭性镰刀菌病的选择之一。

Objective

To investigate clinical features and treatment of hematological malignancies combined with invasive fusariosis in children, and to review the literatures.

Methods

On July 15, 2016, a 13-year-old girl with myelodysplastic syndromes (MDS) and invasive fusariosis who was hospitalized in the Department of Pediatrics, Sun Yat-Sen Memorial Hospital, Sun Yat-Sen University was chosen as study subject, and its clinical features were analyzed. With the following key words of " malignant" " blood system disease" and " invasive fusariosis" as Chinese key words, and " malignant, hematologic disease" and " invasive fuasriosis" as English key words, the literature of hematological malignancies combined with invasive fusariosis were searched from Pubmed database, SinoMed, China National Knowledge Infrastructure (CNKI), Wanfang Data Knowledge Service Platform, and VIP database. Literature retrieval time ranged from Janury 1, 2010 to December 31, 2016, and clinical features of searched literatures were summarized. This study was consistent with the requirements of World Medical Association Declaration of Helsinki revised in 2013.

Results

①This was a case report of MDS with invasive fusariosis based on clinical manifestations and related laboratory findings. After chemotherapy, there were pulmonary infiltrates and limbs, purple-red nodules on the trunk with central necrosis, followed by multiple subcutaneous fluid masses on the extremities. Skin biopsy, bone marrow culture, and mass drainage culture all indicated Fusarium infection. Amphotericin B combined with posaconazole were given to the child. The child was followed up until April 5, 2017, her families gave up treatment and discharged because the primary basic disease could not be relieved. ② The results of setting a retrieval strategy to search relevant literatures showed that a total of 7 domestic and foreign literatures reported a total of 21 children with hematologic malignancies and invasive fusariosis. Among them, 12 were male and 9 were female. The main clinical manifestations were persistent fever, ineffective active anti-infection treatment and poor prognosis. Of the 21 children, 14 died and 7 were cured.

Conclusions

Children with hematological malignancies combined with invasive fusariosis should be diagnosed early and given effective and sufficient antifungal treatment. Amphotericin B combined with posaconazole may be one of the options for the treatment of invasive fusariosis..

图1 患儿(13岁,女性)皮肤硬结活组织检查和培养结果[图1A:真皮深层及皮下脂肪组织内大片陈旧性出血,灶状淋巴细胞、组织细胞浸润(HE染色,低倍);图1B:病灶内见疑似真菌菌丝(HE染色,高倍);图1C:病灶培养出真菌菌丝,符合深部真菌病诊断,有镰刀菌生长(D-PAS染色,高倍)]
图2 患儿(13岁,女性)2016年9月12日进行的胸部CT检查结果(图2A:右侧肺叶、左肺下叶外基底段,可见多发性大片状浸润影;图2B:右侧肺叶、左肺上舌段和左肺下舌段散在结节影和肿物影,可见空气支气管征;图2C:双侧胸腔内见弧状水密度影,右肺上叶及中叶体积明显缩小)
图4 患儿13岁,女性足背近拇趾部一处皮下液性包块
图5 患儿(13岁,女性)2016年11月9日的胸部CT检查结果(图5A:与2016年9月12日的CT检查结果比较,双肺病变范围缩小,双肺尖多发片状浸润影,边界不清;图5B:双肺部分病灶见充气支气管影;图5C:右侧胸腔少量积液,左侧胸腔积液增多)
图6 患儿(13岁,女性)2016年11月29日的胸部CT检查结果(图6A:右侧肺叶、左肺下叶外基底段状浸润影较前2次(2016年9月12日、2016年11月9日)CT检查结果比较,明显吸收;图6B:右侧肺叶、左肺上舌段和左肺下舌段散在结节影和肿物影消失,未见空气支气管征;图6C:双肺病灶较前2次(2016年9月12日、2016年11月9日)CT检查结果比较,吸收好转,病灶范围较前2次缩小)
表1 7篇血液系统恶性疾病合并侵袭性镰刀菌病文献报道的21例患者临床特点
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