Anti-thymocyte globulin combined with cyclosporine A in the treatment of severe aplastic anemia-induced caplillary leak syndrome in children: A case report

doi:10.3969/j.issn.1004-583X.2023.09.008

Abstract

Abstract:

Objective To improve clinicians' understanding of capillary infiltration syndrome (CAS), to reduce missed diagnosis and misdiagnosis, and improve the prognosis. Methods Clinical data of a child with severe aplastic anemia (SAA)-induced CAS treated with anti-thymocyte globulin (ATG) combined with cyclosporine A (CSA) were retrospectively analyzed. The clinical characteristics and treatment course were summarized. Relevant literatures were reviewed as well. Results In this study, we reported a SAA child with CLS on the 13 days of the immunosuppressant treatment. The clinical features included progressive dyspnea, hypotension, peripheral edema, hypoproteinemia, and multiple plasma cavity effusions. After hormonal shock, hydroxyethyl starch expansion, furosemide diuretic dehydration and blood transfusion support, the symptoms and signs were gradually improved. Conclusion CLS is a rare complication during the application of ATG combined with immunosuppressive therapy of CSA in SAA patients, which has the risk of death that should be well concerned. Early recognition, diagnosis and treatment can improve the prognosis.

Key words: anemia, aplastic, antilymphocyte serum, cyclosporine A, capillary leak syndrome

CLC Number:

R556.5

Gu Cuihong, Wang Yang, Lin Li, Wang Lihong, Zhang Zhihua. Anti-thymocyte globulin combined with cyclosporine A in the treatment of severe aplastic anemia-induced caplillary leak syndrome in children: A case report[J]. Clinical Focus, 2023, 38(9): 819-322.

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URL: https://huicui.hebmu.edu.cn/EN/10.3969/j.issn.1004-583X.2023.09.008

https://huicui.hebmu.edu.cn/EN/Y2023/V38/I9/819

References 37

[1]	Zaimoku Y, Patel BA, Shalhoub R, et al. Predicting response of severe aplastic anemia to immunosuppression combined with eltrombopag[J]. Haematologica, 2022, 107(1):126-133. doi: 10.3324/haematol.2021.278413 URL
[2]	Patel BA, Groarke EM, Lotter J, et al. Long-term outcomes in patients with severe aplastic anemia treated with immunosuppression and eltrombopag: A phase 2 study[J]. Blood, 2022, 139(1):34-43. doi: 10.1182/blood.2021012130 URL
[3]	Li R, Zhou J, Liu Z, et al. Predicting response of severe aplastic anemia to rabbit-antithymocyte immunoglobulin based immunosuppressive therapy combined with eltrombopag[J]. Front Immunol, 2022, 13:884312. doi: 10.3389/fimmu.2022.884312 URL
[4]	张凤奎. 我如何治疗难治性重型再生障碍性贫血[J]. 中华血液学杂志, 2020, 41(9):705-709.
[5]	Khan M, Iftikhar R, Chaudhry QU, et al. Response to immunosuppressive therapy in patients of acquired aplastic anaemia: a single center experience from a developing country[J]. J Ayub Med Coll Abbottabad, 2022, 34(Suppl 1)(4):S969-S973.
[6]	Juneja D, Kataria S. Capillary leak syndrome: a rare cause of acute respiratory distress syndrome[J]. World J Clin Cases, 2022, 10(13):4324-4326. doi: 10.12998/wjcc.v10.i13.4324 pmid: 35665129
[7]	中华医学会血液学分会红细胞疾病贫血学组. 再生障碍性贫血诊断与治疗中国专家共识(2017年版)[J]. 中华血液学杂志, 2017, 38(1): 1-5.
[8]	Nejatifar F, Hesni E, Samadani AA. A novel case report of severe aplastic anemia with COVID infection[J]. Ethiop J Health Sci, 2023, 33(1):177-181. doi: 10.4314/ejhs.v33i1.22 pmid: 36890930
[9]	孔令军, 何海龙, 胡绍燕. 猪抗人淋巴细胞球蛋白与兔抗人胸腺细胞球蛋白治疗重型再生障碍性贫血儿童的疗效比较[J]. 中华血液学杂志, 2019, 40(11):953-955.
[10]	Liu L, Miao M, He H, et al. Severe aplastic anemia patients with infection who received an allogeneic hematopoietic stem cell transplantation had a better chance: Long-term outcomes of a multicenter study[J]. Front Immunol, 2022, 13:955095. doi: 10.3389/fimmu.2022.955095 URL
[11]	杨文睿, 张凤奎. 获得性再生障碍性贫血的诊断及治疗[J]. 临床荟萃, 2015, 30(10):1081-1086.
[12]	任蓉, 韩静. 血清G-CSF水平与再生障碍性贫血患儿免疫功能的相关性[J]. 中国实验血液学杂志, 2022, 30(3):819-823.
[13]	Samarasinghe S, Veys P, Vora A, et al. Paediatric amendment toadult BSH Guidelines for aplastic anaemia J[J]. Br J Haematol, 2018, 180(2):201-205. doi: 10.1111/bjh.2018.180.issue-2 URL
[14]	孙悦. 补肾益髓方联合ATG/ALG治疗重型再障疗效分析及从T淋巴细胞阴性选择研究再障发病机制[D]. 济南: 山东中医药大学, 2020.
[15]	董杰. ATG联合CsA治疗重型再生障碍性贫血的临床价值研究[J]. 中国实用医药, 2019, 14(12):127-129.
[16]	杨冰雪, 程单凤, 邱晨希, 等. 环孢素A联合雄激素与单用雄激素治疗国人再生障碍性贫血疗效及安全性分析[J]. 临床荟萃, 2018, 33(10):889-896, 903.
[17]	Bichon A, Bourenne J, Gainnier M, et al. Capillary leak syndrome: state of the art in 2021[J]. Rev Med Interne, 2021, 12, 42(11):789-796.
[18]	Garatti L, Wu MA, Ammirati E, et al. Systemic leak capillary syndrome with myocardial involvement and cardiogenic shock: a case report[J]. Eur Heart J Case Rep, 2022, 6(7): 262.
[19]	Druey KM, Parikh SM. Idiopathic systemic capillary leak syndrome(clarkson disease)[J]. J Allergy Clin Immunol, 2017, 140(3):663-670. doi: S0091-6749(16)32460-5 pmid: 28012935
[20]	马素亭, 孙立荣. 儿童重型再生障碍性贫血并发毛细血管渗漏综合征诊治探讨(附2例报告)[J]. 齐鲁医学杂志, 2012, 27(3):271-272, 274.
[21]	吴传聪, 魏建波, 崔艳铭, 等. 毛细血管渗漏综合征1例[J]. 中华风湿病学杂志, 2022, 26(7):471-473.
[22]	石红妍, 陈延平. 肾综合征出血热生物标志物与病情预测模型研究进展[J]. 传染病信息, 2022, 35(6):486-490.
[23]	潘郭海容, 田圆, 王瀚黎, 等. 脓毒症相关毛细血管渗漏综合征的研究进展[J]. 中国急救医学, 2022, 42(2):163-169.
[24]	梅华鲜, 李永胜, 陈正平, 等. 毛细血管渗漏指数在脓毒症并ARDS早期诊断中的应用[J]. 新医学, 2016, 47(10):681-685.
[25]	Cordemans C, De Laet I, Van Regenmortel N, et al. Fluid management in critically ill patients: the role of extravascular lung water, abdominal hypertension, capillary leak, and fluid balance[J].? Ann Intensive Care, 2012, 2(Suppl 1 Diagnosis and management of intra-abdominal hyperten):S1.
[26]	林柏柏, 钱风华, 钱义明, 等. 脓毒症并发毛细血管渗漏综合征的研究进展[J]. 基础医学与临床, 2022, 42(7):1144-1147.
[27]	印佩如, 倪海滨. 中西医结合治疗脓毒症毛细血管渗漏综合征的研究进展[J]. 中国中医药现代远程教育, 2022, 20(18):206-208.
[28]	廖娜, 张慈婵, 林晓芬, 等. 异基因造血干细胞移植患者预处理后并发毛细血管渗漏综合征的护理[J]. 中华护理杂志, 2019, 54(10):1498-1500. doi: 10.3761/j.issn.0254-1769.2019.10.010
[29]	林柏柏, 沈梦雯, 钱义明, 等. 从“痰饮”论治脓毒症毛细血管渗漏综合征[J]. 陕西中医, 2021, 42(2):222-224, 238.
[30]	沈梦雯, 林柏柏, 钱义明, 等. 四逆升降散对改善脓毒症毛细血管渗漏综合征无创血流动力学指标的效果[J]. 实用临床医药杂志, 2021, 25(14):15-19.
[31]	王宏, 吴锡平, 肖涛, 等. 真武汤治疗重症患者毛细血管渗漏综合征 16 例疗效分析[J]. 大家健康:学术版, 2015, 9(22):103-104.
[32]	黄赟, 王智兰. 参附注射液治疗毛细血管渗漏综合征的临床疗效[J]. 临床合理用药杂志, 2022, 15(18):132-135.
[33]	张立彦, 张利鹏, 王雪芳, 等. 血必净注射液在毛细血管渗漏综合征治疗中的临床研究[J]. 国际检验医学杂志, 2017, 38(7):919-920, 923.
[34]	沈梦雯, 林柏柏, 朱亮, 等. 四逆升降散改善脓毒症毛细血管渗漏综合征的临床研究[J]. 中国中医急症, 2022, 31(3):391-394.
[35]	李智泉, 王晨虹. 云南白药胶囊治疗重度子痫前期毛细血管渗漏综合征[J]. 中西医结合心脑血管病杂志, 2009, 7(7):772-773.
[36]	李智泉. 复方丹参滴丸治疗重度子痫前期合并毛细血管渗漏综合征的临床观察[C]. 复方丹参滴丸美国FDA II期临床试验结果报告暨第四届学术研讨会论文集. 天津: 天津天士力集团, 2010:423-425.
[37]	Qin L, Tan HL, Wang YG, et al. Astragalus membranaceus and salvia miltiorrhiza ameliorate lipopolysaccharide-induced acute lung injury in rats by regulating the toll-like receptor 4/nuclear factor-kappa B signaling pathway[J]. Evid Based Complement Alternat Med, 2018, 2018:3017571.