North East Thames Regional Genetics Service, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
Genetics and Genomic Medicine, University College London Great Ormond Street Institute of Child Health, London, UK.
Health Technol Assess. 2019 Aug;23(46):1-104. doi: 10.3310/hta23460.
Less invasive perinatal and paediatric autopsy methods, such as imaging alongside targeted endoscopy and organ biopsy, may address declining consent rates for traditional autopsy, but their acceptability and accuracy are not known.
The aims of this study were to provide empirical data on the acceptability and likely uptake for different types of autopsy among key stakeholders (study 1); and to analyse existing autopsy data sources to provide estimates of the potential efficacy of less invasive autopsy (LIA) and its projected utility in clinical practice (study 2).
Study 1: this was a mixed-methods study. Parents were involved in research design and interpretation of findings. Substudy 1: a cross-sectional survey of 859 parents who had experienced miscarriage, termination of pregnancy for fetal anomaly, stillbirth, infant or child death, and interviews with 20 responders. Substudy 2: interviews with 25 health professionals and four coroners. Substudy 3: interviews with 16 religious leaders and eight focus groups, with 76 members of the Muslim and Jewish community. Study 2: a retrospective analysis of national data in addition to detailed information from an existing in-house autopsy database of > 5000 clinical cases that had undergone standard autopsy to determine the proportion of cases by clinical indication group for which tissue sampling of specific internal organs significantly contributed to the diagnosis.
Substudy 1: 91% of participants indicated that they would consent to some form of LIA, 54% would consent to standard autopsy, 74% to minimally invasive autopsy (MIA) and 77% to non-invasive autopsy (NIA). Substudy 2: participants viewed LIA as a positive development, but had concerns around the limitations of the technology and de-skilling the workforce. Cost implications, skills and training requirements were identified as implementation challenges. Substudy 3: religious leaders agreed that NIA was religiously permissible, but MIA was considered less acceptable. Community members indicated that they might consent to NIA if the body could be returned for burial within 24 hours. Study 2: in 5-10% of cases of sudden unexplained death in childhood and sudden unexplained death in infants, the final cause of death is determined by routine histological sampling of macroscopically normal organs, predominantly the heart and lungs, and in this group routine histological sampling therefore remains an important aspect of investigation. In contrast, routine histological examination of macroscopically normal organs rarely (< 0.5%) provides the cause of death in fetal cases, making LIA and NIA approaches potentially highly applicable.
A key limitation of the empirical research is that it is hypothetical. Further research is required to determine actual uptake. Furthermore, because of the retrospective nature of the autopsy data set, findings regarding the likely contribution of organ sampling to final diagnosis are based on extrapolation of findings from historical autopsies, and prospective data collection is required to validate the conclusions.
LIA is viable and acceptable (except for unexplained deaths), and likely to increase uptake. Further health economic, performance and implementation studies are required to determine the optimal service configuration required to offer this as routine clinical care.
The National Institute for Health Research Health Technology Assessment programme.
微创围产儿和儿科尸检方法，如影像学检查联合靶向内镜检查和器官活检，可能会解决传统尸检同意率下降的问题，但这些方法的接受程度和准确性尚不清楚。
本研究的目的是为关键利益相关者提供不同类型尸检接受程度的实证数据（研究 1）；并分析现有的尸检数据来源，以提供微创尸检（LIA）的潜在效果的估计及其在临床实践中的预期效用（研究 2）。
研究 1：这是一项混合方法研究。父母参与了研究设计和结果解释。子研究 1：对经历过流产、胎儿异常终止妊娠、死胎、婴儿或儿童死亡的 859 名父母进行横断面调查，并对 20 名应答者进行访谈。子研究 2：对 25 名卫生专业人员和 4 名验尸官进行访谈。子研究 3：对 16 名宗教领袖和 8 个焦点小组进行访谈，其中包括穆斯林和犹太社区的 76 名成员。研究 2：除了对 5000 多例经过标准尸检的临床病例的内部详细信息进行回顾性分析外，还进行了全国性数据的分析，以确定按临床指征组划分的病例比例，其中特定内部器官的组织取样对诊断有显著贡献。
子研究 1：91%的参与者表示他们将同意某种形式的 LIA，54%的人将同意标准尸检，74%的人将同意微创尸检（MIA），77%的人将同意非侵入性尸检（NIA）。子研究 2：参与者认为 LIA 是一个积极的发展，但对技术的局限性和劳动力技能下降表示担忧。成本影响、技能和培训要求被确定为实施挑战。子研究 3：宗教领袖一致认为 NIA 在宗教上是允许的，但 MIA 被认为不太可接受。社区成员表示，如果能在 24 小时内将尸体归还进行埋葬，他们可能会同意 NIA。研究 2：在 5-10%的儿童猝死和婴儿猝死病例中，死因通过对宏观正常器官（主要是心脏和肺部）的常规组织学取样确定，因此，在这组病例中，常规组织学取样仍然是调查的一个重要方面。相比之下，在胎儿病例中，常规的宏观正常器官组织学检查很少（<0.5%）能提供死因，因此 LIA 和 NIA 方法具有潜在的高度适用性。
实证研究的一个主要局限性是它是假设性的。需要进一步研究以确定实际的采用率。此外，由于尸检数据集的回顾性性质，关于器官取样对最终诊断的可能贡献的发现是基于对历史尸检发现的推断，需要前瞻性数据收集来验证结论。
LIA 是可行和可接受的（除了不明原因的死亡），并且可能会增加采用率。需要进一步的健康经济、性能和实施研究来确定提供常规临床护理所需的最佳服务配置。
英国国家卫生研究院卫生技术评估计划。
