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

doi:10.3877/cma.j.issn.1673-5250.2018.04.006

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..

Key words: Hematologic neoplasms, Myelodysplastic syndromes, Fusarium, Biopsy, Antifungal agents, Child

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

文献(第一作者，文献发表年)	年龄(岁)	性别	基础疾病	感染部位	抗真菌治疗方案	结局
Aquino，2010^[2]	18	男	ALL	皮肤	AMB＋VOR	死亡
Aquino，2010^[2]	21	男	ALL	鼻窦	AMB	死亡
Aquino，2010^[2]	81	女	MM	血液系统	CAP＋VOR	治愈
Aquino，2010^[2]	15	女	ALL	皮肤	AMB＋VOR	死亡
Aquino，2010^[2]	38	女	AA	鼻窦	AMB	死亡
Aquino，2010^[2]	57	女	淋巴瘤	血液系统	AMB	死亡
Hsiue，2010 ^[3]	30	男	白血病	肺部	AMB＋FLU	死亡
Hsiue，2010 ^[3]	20	女	白血病	肺部	AMB＋FLU	死亡
Hsiue，2010 ^[3]	45	男	白血病	肺部	AMB＋FLU	死亡
Hsiue，2010 ^[3]	19	女	白血病	血液系统	AMB＋FLU	治愈
Hsiue，2010 ^[3]	56	男	白血病	皮肤	AMB＋VOR	治愈
Hsiue，2010 ^[3]	42	女	淋巴瘤	皮肤	AMB＋VOR	治愈
Hsiue，2010 ^[3]	39	女	MDS	肺部	AMB＋ITR	死亡
谭俊峰，2011 ^[4]	53	女	AML	皮肤	AMB	死亡
Muhammed，2013 ^[5]	59	男	AML	皮肤	AMB＋VOR	死亡
Muhammed，2013 ^[5]	52	男	淋巴瘤	血液系统	AMB＋VOR	死亡
Muhammed，2013 ^[5]	63	男	AML	血液系统	NA	治愈
Muhammed，2013 ^[5]	53	男	AML	血液系统	AMB＋VOR	治愈
黄玲玲，2013 ^[6]	4	男	ALL	皮肤、肺部、血液系统	AMB	治愈
Chan，2014 ^[7]	31	男	ALL	肺部、皮肤	AMB＋VOR	死亡
马永超，2016 ^[8]	22	男	AML	肺部、血液系统	VOR	死亡

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

文献(第一作者，文献发表年)	年龄(岁)	性别	基础疾病	感染部位	抗真菌治疗方案	结局
Aquino，2010^[2]	18	男	ALL	皮肤	AMB＋VOR	死亡
Aquino，2010^[2]	21	男	ALL	鼻窦	AMB	死亡
Aquino，2010^[2]	81	女	MM	血液系统	CAP＋VOR	治愈
Aquino，2010^[2]	15	女	ALL	皮肤	AMB＋VOR	死亡
Aquino，2010^[2]	38	女	AA	鼻窦	AMB	死亡
Aquino，2010^[2]	57	女	淋巴瘤	血液系统	AMB	死亡
Hsiue，2010 ^[3]	30	男	白血病	肺部	AMB＋FLU	死亡
Hsiue，2010 ^[3]	20	女	白血病	肺部	AMB＋FLU	死亡
Hsiue，2010 ^[3]	45	男	白血病	肺部	AMB＋FLU	死亡
Hsiue，2010 ^[3]	19	女	白血病	血液系统	AMB＋FLU	治愈
Hsiue，2010 ^[3]	56	男	白血病	皮肤	AMB＋VOR	治愈
Hsiue，2010 ^[3]	42	女	淋巴瘤	皮肤	AMB＋VOR	治愈
Hsiue，2010 ^[3]	39	女	MDS	肺部	AMB＋ITR	死亡
谭俊峰，2011 ^[4]	53	女	AML	皮肤	AMB	死亡
Muhammed，2013 ^[5]	59	男	AML	皮肤	AMB＋VOR	死亡
Muhammed，2013 ^[5]	52	男	淋巴瘤	血液系统	AMB＋VOR	死亡
Muhammed，2013 ^[5]	63	男	AML	血液系统	NA	治愈
Muhammed，2013 ^[5]	53	男	AML	血液系统	AMB＋VOR	治愈
黄玲玲，2013 ^[6]	4	男	ALL	皮肤、肺部、血液系统	AMB	治愈
Chan，2014 ^[7]	31	男	ALL	肺部、皮肤	AMB＋VOR	死亡
马永超，2016 ^[8]	22	男	AML	肺部、血液系统	VOR	死亡

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