新生儿危重症先天性心脏病适宜筛查窗口期和筛查地点的系统评价和Meta分析

背景 针对新生儿先天性心脏病（CHD）筛查的组配方案的系统评价报道较多,而筛查的最佳窗口期和适宜的筛查机构也同样值得关注。 目的 评估在不同筛查窗口期和筛查机构中进行新生儿危重症CHD（mCHD）筛查的诊断准确性和假阳性率。 设计 诊断准确性研究的Meta分析。 方法 本文文献检索和筛选是在新生儿CHD筛查（NCHDS）指南中实现的。筛查金标准：筛查阳性婴儿行心脏超声检查确诊为mCHD,或随访出现CHD症状和体征后经心脏超声或手术或心脏导管介入术证实为mCHD。 以QUADAS-2量表对纳入文献行偏倚风险及临床适用性评价,提取文献数据,应用随机效应模型汇总敏感度、特异度、假阳性率和假阴性率等诊断准确性参数。 主要结局指标不同筛查窗口期和筛查机构的筛查准确性。 结果 ①关于筛查窗口期的16篇文献中,筛查窗口期在生后≤24 h 9篇, ~48 h 7篇, ~72 h 3篇,6～72 h 3篇,筛查新生儿mCHD的敏感度分别为0.788（95%CI：0.600~0.921）、0.579（95%CI：0.378~0.757）、0.586（95%CI：0.369~0.775）和0.897（95%CI：0.836~0.937）,特异度分别为0.985（95%CI：0.946~0.996）、0.998（95%CI：0.994~0.999）、0.996（95%CI：0.987~0.999）和0.994（95%CI：0.983~0.998）,假阳性率分别为0.015 (95%CI：0.004～0.057)、0.002（95%CI：0.001～0.006）、0.004（95%CI：0.001～0.016）和0.006（95%CI：0.002～0.019）;生后≤24 h、~48 h、～72 h和6～72 h筛查新生儿mCHD的AUC分别为0.916、0.973、0.939和0.973。②关于筛查机构的20篇文献中,产院15篇、NICU 2篇、社区2篇、产院+NICU 1篇,筛查新生儿mCHD的敏感度分别为0.701（95%CI：0.576~0.802）、0.797（95%CI：0.675~0.881）、0.571（95%CI：0.230~0.856）和0.821（95%CI：0.555~0944）;特异度分别为0.995（95%CI：0.990~0.998）、0.885（95%CI：0.778~0944）、0.993（95%CI：0.988~0.996）和0.916（95%CI：0.189~0.998）;产院和NICU筛查新生儿mCHD的假阳性率分别为0006 (95%CI：0.003~0.011)和0.030 (95%CI：0.001~0.636),AUC分别为0.960和0.757;产院生后≤24 h亚组敏感度和特异度分别为0.817和0.996,生后＞24 h亚组敏感度和特异度分别为0.680和0.996。 结论 追求筛查的高敏感度、接受高假阳性率时可以选择生后6~24 h内进行筛查;追求低假阳性率时可选择生后24~72 h之后筛查,应选择在产院开展筛查mCHD,准确性高、假阳性率低。     

应用血肌酸激酶或肌酸激酶MM同工酶筛查新生儿杜氏肌营养不良症的系统评价/Meta分析

背景：目前应用血CK或其同工酶（CK MM）筛查新生儿杜氏肌营养不良症（DMD）的临床研究较少,其准确性尚不明确。 目的：系统评价血CK或CK MM筛查新生儿DMD的准确性。 设计：系统评价/Meta分析。 方法：检索Medline（PubMed）、Embase、Cochrane Library、Web of Science、Scopus、中国知网数据库、中国生物医学文献数据库、万方数据库和维普中文期刊全文数据库,检索起始时间均为1975年1月1日,Medline（PubMed）检索截止时间为2022年11 月5日,其他数据库检索截止时间为2022年10月5日。纳入以血CK或CK MM筛查新生儿DMD的诊断准确性研究。采用QUADAS 2量表对纳入文献进行偏倚风险及临床适用性评价;提取文献数据,应用Stata 15.0和Meta Disc 1.4软件进行Meta分析;合并敏感度、特异度、阳性似然比（PLR）、阴性似然比(NLR)、诊断比值比(DOR),并绘制综合受试者工作特征曲线（SROC）,计算AUC和Q指数值。 主要结局指标：敏感度和特异度。 结果：CK筛查DMD纳入11篇文献,共1 351 953例新生儿;敏感度97%（95%CI:88%～99%）,特异度100%（95%CI:100%～100%）,PLR 1 131（95%CI:370～3 455）,NLR 0.01（95%CI:0.00～0.19）,DOR 16 476 (95%CI:4 115~65 963),AUC为0.995 4,Q指数为0.974 0;Deeks检验P=0.12,发表偏倚的可能较小。CK MM筛查DMD纳入5篇文献,共156 547例新生儿;敏感度和特异度均为100%（95%CI:100%~100%）,PLR 3 925（95%CI: 3 925~3 925）,NLR 0.00（95%CI:0.00~0.00）,DOR 23 094 (95%CI:5 773~92 384);AUC为0.925 2,Q指数为0.859 4。 结论:应用血CK或CK MM筛查新生儿DMD的准确性高,有助于早期诊断DMD。     

吗替麦考酚酯治疗抗髓鞘少突胶质细胞糖蛋白抗体相关疾病疗效的系统评价和Meta分析

背景：目前预防多相性病程的髓鞘少突胶质细胞糖蛋白抗体相关疾病（MOGAD）的复发常用药物较多，但尚无最佳治疗药物推荐。 目的：探讨吗替麦考酚酯（MMF）治疗MOGAD的疗效。 设计：系统评价和Meta分析。 方法：检索英文数据库：PubMed、Embase、Web of Science和Cochrane，中文数据库：万方、中国知网和维普；检索时限均为从建库至2021年6月1日。纳入研究人群为不限性别、年龄、种族且符合MOGAD诊断标准的文献，以 ＭＭF治疗时间≥６个月为干预，结局指标包括复发率或年复发率（ARR）之一的RCT、非RCT、队列研究、病例系列报告（≥3例）和系统评价/Meta分析；删除MMF治疗符合修正治疗定义，无法获得全文，全文非英文或中文及无法提取本文要求的数据信息的文献；提取数据后采用R软件（V4.2.1）进行Meta分析，并对纳入文献行偏倚风险评价。 主要结局指标：无复发率、ARR。 结果：12篇（161例）文献进入本文分析，其中1篇前瞻性队列研究，11篇病例系列报告。儿童57例，成人80例，成人和儿童不能区分24例。起病年龄1~74岁。随访时间为6~67个月。Meta分析结果显示MMF治疗MOGAD的无复发率为74%（95%CI ：58%~89%），各项研究异质性显著（I2=83%，P＜0.01）；MMF治疗MOGAD后的ARR较治疗前的ARR下降了1.38（95%CI：0.94~1.82)次/年，各项研究无显著异质性（I2=0%，P=0.62）。总体不良反应发生率为8%（7/87)，5.7%（5/87）因严重不良反应而中止治疗。 结论：本文研究结果提示，MMF用于MOGAD预防复发的治疗至少6个月，无复发率可达74%，MMF治疗后较治疗前的ARR下降了1.38次/年，尚有待设计更好的干预性研究结果证实。     

系统评价早产对儿童智力发育的影响及早期干预作用

目的：评价早产对儿童智力发育的影响及早期干预的作用。方法：检索Cochrane图书馆、PubMed、EMBASE、万方数据库、中国期刊全文数据库和中国维普数据库,检索起止时间均为1980年1月至2011年11月。由2名系统评价员进行资料提取和质量评价,数据分析采用RevMan 5.0.13软件,检验异质性,并根据异质性结果选择相应的效应模型。结果:共纳入18项研究评价早产对儿童智力发育的影响,15项研究（16篇文献）评价早期干预对早产儿智力心理发育的作用。9篇文献提及随机分配方法,9篇文献提及分配方案隐藏,9篇文献提及对研究对象、治疗方案实施者、研究结果测量者采用盲法,12篇文献提及结果数据的完整性,10篇文献提及选择性报告研究结果,没有文献提及其他偏倚来源。早产儿智商（IQ）明显低于足月儿（SMD=-11.62,95％CI: -13.21~-10.03）,差异有统计学意义（Z=14.37,P<0.001）。对胎龄与IQ的关系进行分层分析,发现胎龄越低,IQ越低,儿童期随访,IQ从胎龄34~36周的112.7逐渐降至胎龄25周的82.1。对早期干预的作用进行了评价,各组婴儿（早产儿纠正年龄）于生后6、12、18、24、36个月采用贝利婴儿发育量表（BSID）测定智力发育指数（MDI）和运动发育指数（PDI）,结果表明早期干预组MDI与PDI均较对照组显著提高,P均<0.01。早期干预可显著降低早产儿智力异常的发生率（OR= 0.25,95％CI: 0.15~0.41）。结论:本Meta分析表明早产儿IQ明显低于足月儿,并随着胎龄的降低而逐渐减低,对早产儿早期干预可明显改善早产儿智力水平,降低智力异常的发生率。但早产儿干预仍缺少长期的随访研究,需要进一步的评价。     

儿童和青少年脑性瘫痪临床实践指南选题的范围综述

背景：儿童和青少年脑瘫的康复期望高质量的临床实践指南,指导康复实践者通过科学合理的评估与干预,改善儿童和青少年脑瘫的健康状态。 目的支持、改进和完善指南核心专家组提出的儿童和青少年脑性瘫痪临床实践指南（CANDLE）选题架构层次。 设计：范围综述。 方法：检索英文数据库（谷歌学术搜索）和中文数据库（中国知网、万方和维普）;灰色文献检索谷歌和百度网站;检索起止日期为2010年1月1日至2022年5月2日。纳入儿童和青少年脑瘫知识合成类文献（指南、共识、系统评价、范围综述）。根据谷歌学术搜索网站默认的智能排序依次筛选,阅读题目和摘要初筛直至连续50篇均无符合纳入标准的文献为止;筛选保留文献下载全文再筛;再筛保留文献的参考文献与保留文献查重,不重复文献行题目和摘要筛选。自制文献数据提取表,提取文献一般特征;范围综述界定问题：《国际功能、残疾和健康分类 儿童青少年》（ICF CY）运用状况,功能是如何被表述的,儿童脑瘫康复的评估、发育、交互等如何被总结归纳的;6名数据提取员采用定性和定量的方法总结结果,CANDLE核心专家成员通过讨论回答范围综述界定的问题。 主要结局指标：指南选题总体框架层次。 结果：372篇文献纳入本文范围综述,指南、共识、系统评价和范围综述均呈现随年份逐步增多趋势,英文范围综述2010至2018年7篇,~2022年42篇。英文文献研究者最关注干预技术、评估技术和功能状态,中文文献中干预技术占比66.7%。181篇英文干预技术文献中,涉及37种技术。整体架构采用了ICF CY或ICF理念4篇,部分采用了为78篇。47篇共识和指南中,9篇共识和7部指南以功能为切入,与运动和姿势相关的功能切入点包括：肌张力、移动、手部活动、继发肌肉骨骼障碍、日常生活活动能力和休闲活动。104篇（31%）文献单独论述评估方法,采用基于共识的健康测量工具遴选标准（COSMIN）方法20篇。9篇（3%）单独阐述发育特性对功能的影响。41篇（12.1%）通过相关性、影响因素等方法分析了各种功能之间以及与背景性因素的交互作用。 结论：范围综述支持、改进和完善了CANDLE总体架构,修正为以功能为切入点建立8个维度,以ICF CY定位、评估、发育、交互性、干预5要素表现维度,在要素下产生选题。     

儿童解没食子酸链球菌脑膜炎系统评价

背景：国内外儿童解没食子酸链球菌（SG）所致脑膜炎报道较少，临床特点尚不明确。 目的：系统评价儿童SG脑膜炎的临床特点。 设计：系统评价。 方法：系统检索PubMed、Embase、Web of Science、中国知网、万方和维普数据库中儿童SG脑膜炎的相关文献，检索时间均为建库至2022年11月9日。纳入报道儿童SG感染且致细菌性脑膜炎的病例报告文献；排除综述、会议论文、仅有摘要不能获取全文的文献、全文非中文和英文的文献。由2位评价员独立筛选文献、提取资料，按照病例报告及病例系列报告质量评价工具表对纳入的文献进行质量评价，提取文献特征、病例临床特征、实验室指标和SG药敏结果。 主要结局指标：存活率和并发症。 结果：17篇文献中的21例SG婴幼儿纳入本文分析，男16例，女4例，1例未报告性别；3例早产儿，16例足月儿，2例未报告胎龄；发病日龄 (14.5±16.6)d；发热14例；神经系统异常17例，有消化道症状9例，有呼吸系统症状9例，心动过速3例。11例报告了CRP检测结果均升高；9例外周血WBC<4×109·L-1，6例＞10×109·L-1。CSF中的WBC计数均升高；18例报告了CSF糖水平，14例<2.2 mmol·L-1；17例报告了CSF蛋白水平，15例＞1 700 mg·L-1。21例CSF培养中20例阳性，18例血培养中16例阳性，5例尿培养中1例阳性，1例粪培养阳性；SG巴氏亚种17例，SG解没食子酸亚种2例，SG 2例。12例单用1种抗生素（11例SG菌株对青霉素/氨苄西林均敏感，1例未报告），9例联用2种抗生素（8例SG菌株对青霉素/氨苄西林敏感，1例耐药），最短抗生素使用时间14 d。6例描述有并发症，其中并发脑室出血和脑室炎各2例，1例合并脑积水、脑室扩张、神经运动迟缓、自主运动不良和肌张力增高，1例同时并发双侧视觉诱发电位下降、硬膜下积液、癫。 结论：SG脑膜炎的发病人群主要为2月龄以下小婴儿，可选择青霉素类药物或头孢噻肟治疗，临床转归大多良好。     

经导管封堵和手术修补继发孔型房间隔缺损对照研究的Meta分析

目的 总结国内、外经导管封堵和手术修补继发孔型房间隔缺损（ASD）对比研究的结果，采用Meta分析法评估经导管封堵ASD的治疗效果，为临床应用提供指导。方法 制定原始文献的纳入标准、排除标准及检索策略，在美国医学索引（Medline）、荷兰医学文摘（EMBASE）药理学分册、中国生物医学文摘（CBMA）及Cochrane 图书馆的临床对照试验资料库（CCTR）内进行相关的对照研究检索，并对文献进行质量评价和资料提取，检索年限均从建库至2006年5月。所有经2种方法治疗ASD的对比研究纳入此项Meta分析，研究指标包括：操作失败率、术后残余分流率和并发症（包括主要并发症和心律失常）发生率等，应用RevMan 4.28软件进行数据处理；计数资料的效应尺度以相对危险度（RR）及其95%CI表示。结果 共检索到相关对照研究文献17篇，排除4篇，符合纳入标准的13篇进入Meta分析，所有研究均为非随机对照试验，未采用分配隐藏和盲法，其中1项研究采用意向性分析。Meta分析结果显示，介入组（n＝1 085）的操作失败率高于手术组（n＝631）（9项对照研究，4.61% vs 0.32％，RR＝5.48，95%CI：2.28～13.21）；术后即时残余分流率介入组（n＝495）低于手术组（n＝182）（2项对照研究，3.84% vs 0，RR＝7.61，95%CI：1.04～55.99），1年后随访的残余分流率介入组（n＝402）和手术组（n＝182）间差异无统计学意义（2项对照研究，1.74% vs 0，RR＝3.54，95%CI：0.45～27.72）；介入组（n＝1 191）并发症的发生率明显低于手术组（n＝845）（13项对照研究，6.80% vs 28.52％，RR＝0.24，95%CI：0.19～0.31），其中主要并发症的发生率（12项对照研究，介入组1 155例，手术组780例；1.56% vs 4.23％，RR＝0.37，95%CI：0.21～0.64）及心律失常发生率（9项对照研究，介入组806例，手术组622例，3.85% vs 11.25％，RR＝0.34，95%CI：0.21～0.53），介入组均显著低于手术组。结论 尽管经导管封堵继发孔型ASD的成功率低于手术修补；但其治疗效果与手术修补相似，且安全性优于手术修补。     

儿童2型糖尿病和糖调节异常筛查方案的研究

目的评价及比较在不同特征人群中进行2型糖尿病（T2DM）和糖调节异常（IGR）筛查的效果及成本，优化和推荐儿童青少年T2DM筛查方案。方法采用分层整群随机抽样方法获取19 593例北京市中小学生，用空腹指末梢血糖（FCBG）对其进行T2DM和IGR筛查，FCBG异常者（≥5.6 mmol·L-1）采用1999年WHO口服葡萄糖耐量试验标准进行诊断。并参照美国糖尿病协会建议儿童青少年T2DM筛查的高危对象标准和Dean提出的诊断标准进行T2DM的分型诊断。将研究人群分为一般人群、超重/肥胖和肥胖组，分别计算回访率、血糖异常阳性率、直接医疗成本和确诊每例成本，根据成本 效果推荐儿童青少年T2DM筛查方案。结果一般人群组，FCBG异常者回访率为47.97%(225/469)，超重/肥胖和肥胖组FCBG异常者回访率分别为59.29%（83/140）和68.00%（51/75），差异有统计学意义。一般人群组T2DM/IGR确诊率最低，为1.67/1 000例；总花费11.21万元和确诊每例T2DM/IGR的花费0.35万元，最高。肥胖组T2DM/IGR确诊率最高，为8.09/1 000例；总花费1.62万元和确诊每例T2DM/IGR的花费0.11万元，最低，较一般人群组分别下降85.55%和68.57%。超重/肥胖组介于两者之间。肥胖、超重/肥胖与一般人群组相比，肥胖组有2例T2DM（33.33%，2/6）漏诊，超重/肥胖组没有T2DM 漏诊。建议在超重/肥胖组中进行T2DM和IGR筛查。进一步选择除超重/肥胖外，与T2DM相关的危险因素（黑棘皮病、T2DM家族史和青春期），随调查对象具有危险因素数量的增加，患病危险性增加，筛查依从性增高，T2DM/IGR确诊率升高，筛查总成本和每例成本降低，但是从调查对象具有3个或以上危险因素开始，对T2DM/IGR筛查敏感度下降，与在一般人群组筛查相比，在4个危险因素人群中进行筛查，将有83.33%的T2DM得不到确诊。结论 本大样本研究以成本 效果分析说明：在儿童中符合健康经济学的T2DM筛查方案是在达到超重标准，同时具有1个以上危险因素（黑棘皮病、T2DM家族史和青春期）儿童青少年中进行目的性筛查。     

C反应蛋白对不明原因发热儿童严重细菌感染诊断价值的系统评价

目的 系统评价CRP对不明原因发热儿童严重细菌感染的诊断价值。方法 检索Medline、EMBASE、Cochrane图书馆、维普中文科技期刊数据库、中国知网、万方数据库和中国生物医学文献数据库，纳入CRP对不明原因发热儿童严重细菌感染诊断价值的文献，检索时间均为建库至2012年10月。采用QUADAS量表对纳入文献进行质量评估，使用MetaDisc 1.4软件对纳入文献进行异质性检验及Meta分析。结果 初检到314篇文献，7篇文献符合纳入标准进入系统评价（n=2 179）。6篇为前瞻性研究，1篇为回顾性研究。CRP的最适界值为30~91 mg·L-1。严重细菌感染的患病率为1.7%~29.3%。4篇文献采用多变量Logistic回归分析显示，CRP是严重细菌感染独立的预测因素。报道敏感度和特异度文献间具有高度的异质性，采用描述性分析，CRP不同最适界值诊断严重细菌感染的敏感度为33%~89%，特异度为75%~97%。报道阳性似然比和阴性似然比的文献间具中度异质性，采用随机效应模式合并，合并阳性似然比为4.43 （95%CI：3.49~5.63），阴性似然比为0.39（95%CI：0.29~0.54）。SROC曲线下面积为0.85，Q指数为0.78。结论 在以不明原因发热就诊的婴幼儿中，CRP对严重细菌感染具有中等的诊断价值，应用时尚需结合临床表现及其他实验室检查指标。     

激素敏感型肾病综合征儿童利妥昔单抗治疗不良事件的系统评价/Meta分析

背景：近20多年来利妥昔单抗（RTX）应用于儿童激素敏感型肾病综合征（SSNS）的治疗较其他免疫抑制剂有更好的疗效，但仍需要积累不良事件的报告情况。 目的：了解儿童SSNS应用RTX治疗后的不良事件。 设计：系统评价/Meta分析。 方法：检索PubMed、Embase、Cochrane、Scopus和中国生物医学文献服务系统数据库，检索时间为建库至2022年6月26日，以SSNS、RTX构建中英文数据库检索式。同一篇文献初筛、全文筛选和证据提取均由2人完成，有争议和不确定的文献由第3人复核。纳入至少1组干预措施使用RTX治疗1~22岁SSNS患儿的研究。 主要结局指标：不良事件发生率。 结果：共纳入47篇文献（中文5篇，英文42篇），7篇双臂干预研究［RCT 5篇，非随机对照试验2篇］和40篇病例系列报告。RTX组较安慰剂或常规免疫抑制剂（对照组）治疗SSNS患儿不良事件发生率（130/184 vs 107/177）、严重不良事件发生率（14/140 vs 9/122）和输液相关反应发生率（19/24 vs 13/24）差异均无统计学意义；感染发生率（56/80 vs 41/62）差异有统计学意义（OR=3.99, 95%CI:1.23~12.97）。RTX治疗SSNS病例系列报告中，不良事件发生率59%（95%CI:55%~63%），严重不良事件发生率7%（95%CI:6%~9%），输液反应发生率31%（95%CI:28%~35%），感染发生率21%（95%CI:18%~24%），血清病发生率5%（95%CI:2%~10%），同时报告外周血中性粒细胞减少和缺乏的研究中，中性粒细胞减少发生率9%（95%CI:5%~17%）、缺乏发生率4%（95%CI:2%~10%），针对低IgG血症研究的低IgG血症发生率为51%（95%CI:42%~60%）。 结论：RTX治疗SSNS未增加不良事件、严重不良事件和感染的发生率，低IgG血症和血清病值得关注。     

Cost of neonatal intensive care delivered through district level public hospitals in India

Objective

To assess the unit cost of level II neonatal intensive care treatment delivered through public hospitals and its fiscal implications in India.

Design

Cost analysis study.

Setting

Four Special Care Newborn Units (SCNUs) in public sector district hospitals in three Indian states, i.e. Bihar, Madhya Pradesh and Orissa, for the period 2010.

Methods

Bottom-up economic costing methodology was adopted. Health system resources, i.e. capital, equipment, drugs and consumables, non-consumables, referral and overheads, utilized to treat all neonates during 2010 were elicited. Additionally, 360 randomly selected treatment files of neonates were screened to estimate direct out-of-pocket (OOP) expenditure borne by the patients. In order to account for variability in prices and other parameters, we undertook a univariate sensitivity analysis.

Main Outcome Measures

Unit cost was computed as INR (Indian national rupees) per neonate treated and INR per bed-day treatment in SCNU. Standardized costs per neonate treatment and per bed day were estimated to incorporate the variation in bed occupancy rates across the sites.

Results

Overall, SCNU neonatal treatment costs the Government INR 4581 (USD 101.8) and INR 818 (USD 18.2) per neonate treatment and per bed-day treatment, respectively. Standardized treatment costs were estimated to be INR 5090 (USD 113.1) per neonate and INR 909 (USD 20.2) per bed-day treatment. In the event of entire direct medical expenditure being borne by the health system, we found cost of SCNU treatment as INR 4976 (USD 110.6) per neonate and INR 889 (USD 19.8) per bed-day.

Conclusions

Level II neonatal intensive care at SCNUs is cost intensive. Rational use of SCNU services by targeting its utilization for the very low birth weight neonates and maintenance of community based home-based newborn care is required. Further research is required on cost-effectiveness of level II neonatal intensive care against routine pediatric ward care.     

Cost-effectiveness and implications of newborn screening for prolongation of QT interval for the prevention of sudden infant death syndrome

OBJECTIVE: To determine the cost-effectiveness of universal and high-risk neonatal electrocardiographic (ECG) screening for QT prolongation as a predictor of sudden infant death syndrome (SIDS) risk in a theoretical group of neonates.Study design: Incremental cost-effectiveness analysis with decision analytic modeling. A hypothetical cohort of healthy, term infants was modeled, comparing options of no screening, high-risk neonate screening, and universal screening. The high-risk strategy is speculative, because no currently accepted methodology is known for identifying infants at high risk for SIDS. Given the uncertain mechanisms of association between prolonged corrected QT interval (QTc) and SIDS, analyses were repeated under different assumptions. Sensitivity analyses were also performed on all input variables for both costs and effectiveness. RESULTS: Under the assumption that neonatal electrocardiographic screening detects long QT syndrome responsive to conventional therapy, the cost-effectiveness of high-risk screening was $3403 per life year gained, whereas universal screening cost $18,465 per additional life year gained. However, if the effectiveness of SIDS therapy falls below 10%, the cost-effectiveness deteriorates to $28,376 per life year saved for the high-risk strategy and $118,900 for universal screening. The analyses were robust to a broad array of sensitivity analyses. CONCLUSIONS: The acceptability of the cost-effectiveness of neonatal electrocardiographic screening is heavily dependent on the pathophysiologic mechanism of SIDS and on the efficacy of monitoring and antiarrhythmic treatment. The nature of this association must be elucidated before routine neonatal electrocardiographic screening is warranted.     

Costing model for neonatal screening and diagnosis of haemoglobinopathies

AIM: To compare the costs and cost effectiveness of universal and targeted screening for the haemoglobinopathies; to compare the cost of two laboratory methods; and to estimate the cost effectiveness of programmes at different levels of prevalence and mix of haemoglobinopathy traits. METHODS: A retrospective review of laboratory and follow up records to establish workload and costs, and estimation of costs in a range of circumstances was made in a haematology department and sickle cell and thalassaemia centre, providing antenatal and neonatal screening programmes in Inner London. The costs for 47,948 babies, screened during 1994, of whom 25 had clinically significant haemoglobinopathies and 704 had haemoglobinopathy traits, were retrospectively assessed. RESULTS: The average cost per baby tested (isoelectric focusing and high power liquid chromatography) was 3.51 Pounds /3.83 Pounds respectively; the cost per case of sickle cell disease identified (IEF/HPLC) was 6738 Pounds /7355 Pounds; the cost per trait identified (IEF/HPLC) was 234 Pounds /255 Pounds; the cost per extra case of SCD and trait identified by universal programme varied. CONCLUSIONS: IEF and HPLC are very similar in terms of average cost per test. At 16 traits/1000 and 0.5 SCD/1000 there was no significant identification cost difference between universal and targeted programmes. Below this prevalence, a targeted programme is cheaper but likely to miss cases of SCD. If targeted programmes were 90-99% effective, universal programmes would cease to be good value except at very high prevalence. Greater use of prenatal diagnosis, resulting in termination, and therefore fewer affected births, reduces the cost effectiveness of universal screening. Screening services should aim to cover a screened population which will generate a workload over 25,000 births a year, and preferably over 40,000.     

Nonmedical costs associated with rotavirus disease requiring hospitalization

BACKGROUND: As the most common cause of severe diarrhea among children, rotavirus has a significant economic impact. Previous studies focused on the direct medical costs of rotavirus infections; however, nonmedical costs account for the majority of the financial burden from this disease. Herein, we report the results from the largest prospective study in the United States determining the nonmedical costs of severe rotavirus infections. METHODS: Prospective, active, gastroenteritis case surveillance was conducted between November 1997 and December 1999 at 3 pediatric medical centers. Rotavirus infection was identified for 548 children admitted between 2 weeks and 5 years of age. Detailed information about nonmedical costs during the prehospitalization, hospitalization and posthospitalization periods was obtained through interviews. RESULTS: The average nonmedical cost per case of rotavirus disease was USD $448.77, including $359.04 for missed work, $56.66 for transportation, $11.90 for oral rehydration solutions, $9.59 for diapers, $6.83 for child care changes, $3.82 for special foods and $0.93 for formula changes. More than one-half of these expenses (53%) occurred outside the hospitalization period, and 80% of the cost was attributable to missed work. CONCLUSIONS: With an estimated 50,000 hospitalizations attributable to rotavirus each year in the United States, the nonmedical costs of severe rotavirus infections may exceed USD $22 million annually. Previous cost effectiveness analyses of rotavirus vaccines substantially underestimated this burden, suggesting that the nonmedical costs associated with mild to moderate rotavirus disease have been similarly underestimated. These findings are needed to assess accurately the cost effectiveness of future rotavirus immunization strategies.     

Cost effectiveness of prevention and treatment of neonatal respiratory distress (RDS) with exogenous surfactant: what has changed in the last three decades?

In 1993, exogenous surfactant products were emerging as licensed treatments for respiratory distress syndrome (RDS), a leading cause of death in preterm newborn babies. Models of cost-effectiveness of alternative treatments showed surfactant to be an expensive but effective and also cost effective treatment. However the most efficient policy for use of surfactant depended on other parallel 'technologies' such as giving antenatal corticosteroids where preterm delivery is anticipated. Following introduction into clinical practice, questions changed from whether to use surfactant, to when, and which product to use. The early models of cost effectiveness were dependent on the neonatal technology in use, and on the costs of neonatal care and prices of surfactant at the time. Little information was available about long term outcomes. The aims of this paper are to summarise the role of surfactant in the economics of newborn care since the late 1980s; to observe the value of studies published in the early 1990s for current decisions; and to comment on recent and possible future economic studies of neonatal surfactant.     

Universal versus targeted screening of infants for sickle cell disease: a cost-effectiveness analysis

OBJECTIVE: To compare the health outcomes, costs, and incremental cost-effectiveness of universal neonatal screening for sickle cell disease (SCD) with screening targeted to African Americans. STUDY DESIGN: A cost-effectiveness analysis was done by using a Markov simulation model that considered the costs and outcomes associated with the prevention and treatment of sepsis in those with sickle cell anemia and sickle beta(0)-thalassemia. Three strategies were compared: (1) no screening, (2) targeted screening of African Americans, and (3) universal screening for SCD. RESULTS: In the base case analysis, targeted screening of African Americans compared with no screening cost $6709 per additional year of life saved, and universal screening compared with targeted screening cost $30,760 per additional year of life saved. In a sensitivity analysis, the cost per additional year of life saved with universal screening compared with targeted screening was positively correlated with the delivery rate of targeted screening and was inversely related to the proportion of African Americans in the population. CONCLUSIONS: Targeted screening of African American newborns for SCD compared with no screening is always cost-effective. Universal screening compared with targeted screening always identifies more infants with disease, prevents more deaths, and is cost-effective given certain delivery rates for targeted screening and proportions of African Americans in the population.     

The costs of early hearing screening in England and Wales

A survey was carried out in 10 centres in England and Wales to determine the costs of hearing screening in the first year of life. The screens that were studied were targeted neonatal, universal neonatal, and the health visitor distraction test (HVDT) or alternative surveillance. Valid data were available from five centres for targeted neonatal screening (TNS), three for universal neonatal screening (UNS), and nine for the HVDT, although only five of the HVDT screens had valid data for follow up costs. The neonatal costs were consistent across the centres surveyed, whereas those for the HVDT screen varied considerably. The mean service costs for TNS, UNS, and the HVDT at 1994 prices were 5052 Pounds, 13,881 Pounds, and 24,519 Pounds for a standardised district of 1000 live births. Three conclusions seem justified. Firstly, UNS need not be prohibitively expensive as it costs considerably less than HVDT screening. Secondly, TNS appears to be a relatively inexpensive way of improving the age of identification of a proportion of the congenitally hearing impaired. Thirdly, given the published yields for UNS and the HVDT, the results indicate that UNS offers the most cost effective overall approach with alternative systems in place to identify late onset permanent hearing losses.     

Fundamentals of clinical economics in health interventions

Health economics is a science of efficient use of medical resources. Economic evaluation deals with both inputs or costs and outputs or consequences and is done when there are choices. The inputs or costs can be direct, indirect or intengible. The consequences can be measured as natural health units or utilities which aggregate multiple health units or quality adjusted life years. In the current health care scenario in India the cost data is usually lacking. An exact way for estimating the cost is elicitation of opportunity cost which is the value of time or input at its highest. The total cost is, the cost of producing a particular quantity of the output and is made up of fixed and variable costs. Fixed costs do not vary with the quantity of output but variable costs do. In India, the total cost of health care provided by a multidisciplinary team can be estimated either by the ‘per diem’ approach which assumes no cost sharing by the departments, or by the allocation method which takes into account the quantity of service provided by each department and costs them individually. Cost effectiveness or utility and cost-benefit analysis are considered complete economic evaluation. Analyses are done from a pre-specified perspective which could be patients’, payers’, providers’ or societal. Costs incurred at a later time are discounted. Such evaluations help us make informed decision to choose between various health interventions. Their increasing use in health research in the developing countries is needed.     

肺脏超声对新生儿肺炎诊断价值的Meta分析

目的　采用 Meta 分析方法来评价肺脏超声对新生儿肺炎的诊断价值。方法　检索PubMed、Cochrane library、Embase、CNKI 和万方数据库,按照诊断性试验的纳入和排除标准,获取符合本研究要求的文献,检索时间为建库至 2018年9月。共检索到907篇文献,选取其中8篇进一步分析,并对文献质量进行QUADAS2标准评价,采用Q检验和I 2统计量进行异质性检验,采用 Meta-Disc 1.4 软件进行统计学分析,评价肺脏超声对新生儿肺炎的诊断价值。 结果　共纳入8篇文献、1078例。采用随机效应模型对纳入的研究进行统计分析,合并敏感度为0.96（95%CI：0.95～0.98）,合并特异度为0.98（95%CI：0.95～0.99）,合并阳性似然比为19.52（95%CI：5.00～76.15）,合并阴性似然比为0.04（95%CI：0.01～0.16）,合并诊断优势比为565.45（81.80～3908.58）,拟合ROC 曲线下面积0.9950。结论　肺脏超声诊断新生儿肺炎的敏感度和特异度较X线高。鉴于其相对容易操作、实时可用、低成本和无电离辐射,目前的证据支持超声作为新生儿肺炎诊断的替代手段。