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Minimally invasive autopsy for fetuses and children based on a combination of post-mortem MRI and endoscopic examination: a feasibility study.
Health Technol Assess 2019; 23(46):1-104HT

Abstract

BACKGROUND

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.

OBJECTIVES

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).

REVIEW METHODS

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.

RESULTS

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.

LIMITATIONS

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.

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.

FUNDING

The National Institute for Health Research Health Technology Assessment programme.

Authors+Show Affiliations

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.Department of Histopathology, Great Ormond Street Hospital for Children NHS Foundation Trust and Institute of Child Health/University College London, London, UK.Department of Psychological Services, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.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.Department of Radiology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.Antenatal Results and Choices, London, UK.Institute of Child Health; Population, Policy and Practice, University College London, London, UK.Comprehensive Clinical Trials Unit, University College London, London, UK.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.Department of Histopathology, Great Ormond Street Hospital for Children NHS Foundation Trust and Institute of Child Health/University College London, London, UK.

Pub Type(s)

Journal Article

Language

eng

PubMed ID

31461397

Citation

Lewis, Celine, et al. "Minimally Invasive Autopsy for Fetuses and Children Based On a Combination of Post-mortem MRI and Endoscopic Examination: a Feasibility Study." Health Technology Assessment (Winchester, England), vol. 23, no. 46, 2019, pp. 1-104.
Lewis C, Hutchinson JC, Riddington M, et al. Minimally invasive autopsy for fetuses and children based on a combination of post-mortem MRI and endoscopic examination: a feasibility study. Health Technol Assess. 2019;23(46):1-104.
Lewis, C., Hutchinson, J. C., Riddington, M., Hill, M., Arthurs, O. J., Fisher, J., ... Sebire, N. J. (2019). Minimally invasive autopsy for fetuses and children based on a combination of post-mortem MRI and endoscopic examination: a feasibility study. Health Technology Assessment (Winchester, England), 23(46), pp. 1-104. doi:10.3310/hta23460.
Lewis C, et al. Minimally Invasive Autopsy for Fetuses and Children Based On a Combination of Post-mortem MRI and Endoscopic Examination: a Feasibility Study. Health Technol Assess. 2019;23(46):1-104. PubMed PMID: 31461397.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Minimally invasive autopsy for fetuses and children based on a combination of post-mortem MRI and endoscopic examination: a feasibility study. AU - Lewis,Celine, AU - Hutchinson,John C, AU - Riddington,Megan, AU - Hill,Melissa, AU - Arthurs,Owen J, AU - Fisher,Jane, AU - Wade,Angie, AU - Doré,Caroline J, AU - Chitty,Lyn S, AU - Sebire,Neil J, PY - 2019/8/29/entrez PY - 2019/8/29/pubmed PY - 2019/8/29/medline KW - AUTOPSY KW - BIOPSY KW - CAUSE OF DEATH KW - CONSENT KW - CORONERS KW - FETAL KW - HEALTH PROFESSIONALS KW - LAPAROSCOPIC KW - MRI KW - PAEDIATRIC KW - PARENTS KW - PERINATAL SP - 1 EP - 104 JF - Health technology assessment (Winchester, England) JO - Health Technol Assess VL - 23 IS - 46 N2 - BACKGROUND: 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. OBJECTIVES: 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). REVIEW METHODS: 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. RESULTS: 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. LIMITATIONS: 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. 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. FUNDING: The National Institute for Health Research Health Technology Assessment programme. SN - 2046-4924 UR - https://www.unboundmedicine.com/medline/citation/31461397/Minimally_invasive_autopsy_for_fetuses_and_children_based_on_a_combination_of_post_mortem_MRI_and_endoscopic_examination:_a_feasibility_study_ L2 - https://doi.org/10.3310/hta23460 DB - PRIME DP - Unbound Medicine ER -