Article Text

PDF

Prospective parental consent for autopsy research following sudden unexpected childhood deaths: a successful model
  1. S Thayyil1,
  2. N J Robertson2,
  3. A Scales3,
  4. M A Weber4,
  5. T S Jacques5,
  6. N J Sebire6,
  7. A M Taylor7
  1. 1
    UCL Institute of Child Health, London, UK
  2. 2
    UCL Institute of Women’s Health and University College London Hospitals, London, UK
  3. 3
    Paediatric Intensive Care Unit, Great Ormond Street Hospital for Children, London, UK
  4. 4
    Great Ormond Street Hospital for Children, London, UK
  5. 5
    Neural Development Unit, UCL Institute of Child Health and Great Ormond Street Hospital for Children, London, UK
  6. 6
    Department of Histopathology, Great Ormond Street Hospital for Children and UCL Institute of Child Health, London, UK
  7. 7
    Cardiovascular Unit, UCL Institute of Child Health and Great Ormond Street Hospital for Children, London, UK
  1. Dr Sudhin Thayyil, Centre for Cardiovascular Imaging, UCL Institute of Child Health and Great Ormond Street Hospital for Children, Great Ormond Street, London WC1N 3JH, UK; s.thayyil{at}ich.ucl.ac.uk

Abstract

Background: Organ retention issues, recent changes in the Coroners’ (Amendment) Rules 2005 and the Human Tissue Act have resulted in pessimism regarding prospective consent for paediatric autopsy research in the UK.

Objectives: To examine the feasibility and acceptability of a prospective telephone consenting model for post-mortem magnetic resonance (MR) imaging research in HM Coroners’ cases.

Design: Following each autopsy referral from the HM Coroner, permission to contact the family for research was requested. A family liaison sister, with experience in dealing with bereaved families, then contacted the parents by telephone, explained the study and obtained oral, and then written consent for post-mortem imaging.

Setting: London and an area south of London.

Results: Of 76 eligible HM Coroners’ cases referred during the study period, permission to contact parents (provided by the HM Coroners’ Office) was obtained for only 32 cases (42%). The research sister contacted 32 parents during the study period of whom 31 (96.8%) gave oral research consent. “Helping other parents in the future” and “the importance of post-mortem research” were the main reasons for parents wanting to participate in research.

Conclusions: Prospective consenting for HM Coroners’ cases for research is feasible in children, and can be done ethically by parental consenting via telephone contact before autopsy by appropriately trained staff. However, close co-ordination between mortuary staff, HM Coroners, research staff and medical staff is required. This model may be useful in performing post-mortem research in HM Coroners’ cases and in developing paediatric tissue and brain banks in the UK.

Statistics from Altmetric.com

The vast majority of paediatric, as opposed to fetal and perinatal, autopsies in the UK are performed on behalf of HM Coroners, since hospital post-mortems with consent are now uncommon. Thus, many aspects of post-mortem research depend primarily on successful recruitment of HM Coroners’ cases.1 Such research is crucial especially in cases of sudden and unexpected infant deaths (SUDI), where despite detailed post-mortem examination by specialist paediatric pathologists, more than 60% of cases remain unexplained.2 3

Controversies relating to organ retention and recent changes to the Coroners’ Rules and the Human Tissue Act in the UK have added to the complexities of autopsy research in children.1 While HM Coroners do not require parental consent for retention of tissue for diagnostic investigation of the cause of death, no tissue or data (eg, magnetic resonance (MR) imaging data) can be collected or retained for research purposes alone, without explicit parental consent. According to the new regulations, it is the duty of the HM Coroner to inform the pathologist of the parents’ wishes regarding the use of residual tissue in research within a specified time period. However, many HM Coroner’s Officers are not trained to, nor have adequate resources to, obtain informed research consent from parents. Nevertheless, if parental wishes are not known within this period, the tissues taken at autopsy must be disposed of by the hospital. HM Coroners vary widely in their adoption and interpretation of the new rules4 and only in a minority of cases is the appropriate parental consent information obtained and given to the pathologist.5 6

Moreover, even in cases where HM Coroner’s Officers do provide signed consent forms, these are usually not available at the time of autopsy.5 Most prospective autopsy research requires specimens to be taken soon after death or during the autopsy; delay or improper processing will diminish or even abolish their value for research purposes.7 8

The major issues discussed during the organ retention debate were lack of appropriate consent and involvement of the next of kin. Many affected families stated that they had no objection to post-mortem research and retention of tissues for same, their complaint was primarily that they were neither aware of nor involved in such a process.8 However, contacting newly bereaved parents for research consent has been perceived as unethical7 911 and this complex ethical situation has led to the virtual disappearance of prospective autopsy research in the UK.

As part of a large Department of Health (DH) study on “less invasive autopsy” by post-mortem MR imaging (MaRIAS: Magnetic Resonance Imaging Autopsy Study) in 600 fetuses, newborns and children,12 we aimed to perform non-invasive post-mortem MR and CT imaging investigations in HM Coroners’ cases and therefore wanted to explore the feasibility and acceptability of obtaining parental consent prior to autopsy using a telephone consenting model.

METHODS

Prior to the start of the study, 20 key HM Coroners from London and the surrounding areas (out of a total of 65 HM Coroners referring cases to Great Ormond Street Hospital for Children) were approached, the study was explained and assistance with this process was requested. It was made clear that MR and CT scans would be performed without extra cost to HM Coroners and would not cause any delay to conventional autopsy. The study was approved by the local research ethics committee and was funded by the UK Department of Health.

This study was performed over a 5-month period between February and July 2008. Following each referral for an HM Coroner’s autopsy, a researcher contacted the HM Coroner’s Officer, requesting permission to contact the parents (fig 1). If agreed, a senior family liaison sister (trained in family counselling and with extensive experience in dealing with recently bereaved families) then contacted the parents by telephone, explained the study and took oral consent for MR imaging.

Figure 1

Flow chart for the consenting process. GOSH, Great Ormond Street Hospital for Children.

A written consent form was then sent through the post. Responses of the parents and HM Coroner’s Officers regarding participation in the research were recorded in a systematic way. MR and CT imaging were performed prior to a standard HM Coroner’s autopsy in cases where consent was obtained.

RESULTS

Of the 20 HM Coroners approached for assistance prior to the start of the study, two declined, one requested additional funding to participate, two agreed and 15 did not reply.

During the study period, 76 cases from 38 different HM Coroners were eligible for recruitment. HM Coroner’s Officers were approached in all of these cases and permission was requested to contact the parents for research, except in those cases where the Coroner had requested no future contact, or if the autopsy had to be carried out within 4 h of arrival. Permission to contact the parents was granted in 32 cases.

The research nurse contacted all 32 parents by telephone and 31 (96.8%) gave oral research consent. The median age at death was 2 months (range 1 day to 7 years) and the median time after death at the time of consenting was 2 days (1–7 days). The median length of telephone conversations was 25 min (5–90 min). In three cases, the parents requested more time to consider their options and requested a second telephone call some hours later; all three parents consented following the second telephone call.

Responses of the HM Coroner’s Officers and parents are given in tables 1 and 2, respectively. All parents felt that telephone contact by the bereavement nurse was an appropriate way of requesting consent, including the parent who refused consent to participate in the research. All the parents wanted to know more about the post-mortem process, when it would take place and when the body would be returned to them. Most parents spontaneously talked about the circumstances that led to the death of their child. The script used by the family liaison sister when seeking consent is given in appendix 1.

Table 1 Responses from the HM Coroners/Coroner’s Officers regarding contacting parents for research
Table 2 Reason given by parents for agreeing or declining to participate in the research

An information leaflet (appendix 2) and a written consent form (appendix 3) were sent out in the post and were received back between 1 and 8 weeks after telephone consenting in all cases, without any reminders. Two parents requested that the research papers published from the study be sent to them. Three parents contacted the research doctor directly at a later stage by telephone to make sure that the child had indeed been recruited into the study.

DISCUSSION

We report, for the first time, that prospective parental consent for research prior to autopsy is feasible following the unexpected death of a child and can be undertaken in an ethical manner that appears acceptable to the majority of parents. Contrary to popular belief that newly bereaved parents would react angrily to such an approach, we found that most parents viewed the process positively and did not have any objections to the telephone approach. Indeed, most parents spontaneously stated their wish to help other parents in similar situations in the future as their main reason for participating in the study. This model has major implications for paediatric autopsy research in the UK.

The model in this study is very similar to the prospective telephone consenting model that was used in Edinburgh to establish MRC Sudden Death Brain and Tissue Bank in adults.8 9 This MRC tissue bank was established by a joint effort with the area Procurator Fiscal Service (analogous the HM Coroner in England and Wales) in Edinburgh, the police and forensic pathologists. The model was highly successful in contributing to a European network (BrainNet Europe) for brain tissue in adults.8 10 Although some of our initial approaches to the HM Coroners in London and the surrounding areas were unsuccessful, as the study progressed the majority of HM Coroners became supportive of the research.

It needs to be appreciated that the sudden unexplained death of a previously healthy child is a traumatic event which may have very different implications for the family compared to the death of an elderly relative. Therefore, it cannot be assumed that a model that works for adults and the elderly is directly applicable to paediatric cases. In California, legislation was recently amended for mandatory inclusion of SUDI cases for research without parental consent.7 Several experts have proposed a similar model for other countries, making research use of post-mortem tissue “opted in”, for the greater benefits of society;7 however, this was rejected by the UK parliament.

Consenting by telephone has been suggested to be intrusive as opposed to consent request by post. Again, in this small cohort, we did not find any parent who found this method inappropriate when specifically asked. In fact, it appears that the opportunity to discuss the post-mortem process soon after death with a bereavement nurse was beneficial to parents, although the nurse did not offer specific bereavement counselling. Clearly the experience and personality of the person taking research consent is of paramount importance.

The perception of professionals that parents are unwilling to consent to research in HM Coroners’ post-mortem cases (the HM Coroners only gave permission for 42% of parents to be contacted by telephone) appears to differ from the parents’ willingness to take part in research (97% of those subsequently asked). Although this may represent extremely good screening by the coroners (in that only those who were referred would agree to take part in the study), it is more likely that the preconceived opinions of professionals involved in the post-mortem process may be different from the actual feelings of most parents regarding participation in research.

Snowdon et al have reported a sharp contrast between the attitudes of clinicians and bereaved parents to participation in post-mortem research.11 13 Post-mortem research was highly valued by parents who were deeply affected by the loss of their baby. Parents were keen to make a contribution to research, which was driven primarily by altruism and a feeling that the child’s life was not wasted. Such participation is thought to be beneficial in the bereavement process. Conversely, clinicians may be reluctant to recruit children in trials related to post-mortem studies, for the fear of seemingly inappropriate and insensitive requests. The authors comment that the caution and selectivity exercised by the doctors may not be appropriate, at least for some parents.

Traditionally autopsy research in the UK has been performed by obtaining a waiver for explicit parental consent from the host institutional research ethics committees, or by obtaining retrospective parental consent at a later stage.1416 In HM Coroners’ cases, even though the coroner has the authority to permit any post-mortem investigation into the cause of death, this cannot be used for post-mortem research studies, which involve additional investigations or sample taking. Prospective parental consent is required prior to performing any additional research investigation in such cases. Ethical concerns about such research have been raised even for non-invasive post-mortem research (eg, MR imaging), when performed without explicit parental consent.14 17

What is already known on this topic

  • Changes to the Human Tissue Act and an amendment to the Coroners’ Rules have affected autopsy research following infant deaths.

  • Attitudes of newly bereaved parents towards post-mortem research differ from that of professionals.

What this study adds

Parental research consenting before autopsy using a family liaison nurse can be undertaken in an ethical manner that appears acceptable to the majority of parents.

Engagement of parents is the key to credible post-mortem research, ensuring that lessons are learnt from the organ retention discussions. The proposed “Less Invasive Autopsy” project by the Department of Health is based on such parental engagement, and is intended to eventually offer a modified autopsy protocol, which, along with continued involvement of parents, will hopefully improve the consented autopsy rates in hospital deaths of all ages as well.

The model we have proposed could also potentially be used to allow the creation of post-mortem research tissue banks that could be used to address the continuing issues related to sudden death research. However, at present this system is far less effective than the consenting system used by the MRC Sudden Death Brain and Tissue Bank in collaboration with the Procurator Fiscals in Scotland.8 10 This is because of regional differences in coroners’ rules, so an amendment to HM Coroners’ rules would be required to establish a similar system in England.4 5 From a pragmatic perspective, we would propose a model for discussing all areas of research in which the HM Coroner’s Officer provides the hospital with parent contact details where appropriate at the time of referral and a bereavement nurse then contacts the family to explain the autopsy process and the expected time frames and takes oral consent for post-mortem research, such as imaging and tissue retention, prior to autopsy, which would be followed up by written consent. This model may also be acceptable and beneficial to the under-resourced HM Coroners’ system in the long term.

While the storage of tissues from consented cases would be covered by the ethics approval received by individual projects, collaborative research would be supported by the establishment of paediatric post-mortem tissue banks, and use of this model may be the first step towards such a process. It is likely that even in hospital cases, more parents may consent to the use of post-mortem tissues for research if an appropriate bereavement nurse is involved in such discussions.

LIMITATIONS

Our institution has specialist paediatric pathologists and a full time paediatric neuropathologist, and undertakes a large number of sudden infant death autopsies, so it could be argued that there may be a potential selection bias in that referrals may involve more complex cases and therefore parents may be more willing to participate in post-mortem research. However, both perinatal and paediatric autopsies in the UK are now being progressively undertaken in selected tertiary referral centres and we feel that this model could be useful in all such centres. Moreover, all these centres would have a family liaison and/or bereavement nurse attached to intensive care units, who could be utilised for consenting purposes.

CONCLUSION

We conclude that prospective parental consenting by telephone is feasible before paediatric autopsies in HM Coroners’ cases. However, the current system allows this to occur only in a minority of cases and therefore many parents may be denied the opportunity to participate in post-mortem research. We urge policy makers, HM Coroners, pathologists and funding bodies to join forces to develop such models for future post-mortem research in the UK.

Acknowledgments

We are indebted to Dr Alistair Parker, who has guided us through the process of this post-mortem project, Joyce Epstein (Foundation for the Study of Infant Deaths, FSID) for advising us regarding the recruitment of SUDI cases and Professor Jeanne Bell (Professor of Neuropathology, University of Edinburgh) for advice regarding the consenting model.

Appendix 1

Script used by the family liaison sister for consenting

  1. General introductions.

  2. Confirm that parents are aware of the autopsy being performed at Great Ormond Street Hospital for Children.

  3. Sympathetic listening to parents if they wish to talk spontaneously about events around death.

  4. Answer parental queries, if any, regarding post-mortem examination, time when it will be performed and when the child would be released for burial/cremation.

  5. Explain regarding the research on post-mortem MR imaging.

  6. Check if parents would like to have some time to think about it. (In which case a second telephone call is made if parents wish.)

  7. Obtain oral consent if parents wish to participate.

  8. If parents do not wish to participate, express thanks and re-assure their wishes will be honoured.

  9. Check if the parents considered the cold call obtrusive and apologise if that was the case.

  10. Send the information leaflet and consent form by post for parents to sign and return.

Appendix 2

VERSION X

Date

Researchers: Dr XXXXXXXX

Post-mortem magnetic resonance imaging in fetuses, newborn and children: a comparative study with conventional autopsy

Information leaflet for parents

Thank you for taking the time to read this leaflet. We know that this is a difficult time for you and appreciate the time you are taking to read this leaflet.

Background to the study

MRI (magnetic resonance imaging) and CT scans, as you may be aware, are special techniques to get images of the body. An MRI scan can examine internal organs in detail and may be able to identify some of the problems that can be detected by a post-mortem examination. In some cases, we believe that MRI may even be better than a post-mortem examination. Many parents are understandably upset about the thought of their baby undergoing a post-mortem. We are doing this study to find out if an MRI scan of the whole body can give similar information to that of a post-mortem, so that in future we might be able to offer an MRI scan instead of a post-mortem.

What will happen if we agree to take part?

If you agree to take part we will arrange for your baby to have an MRI scan (and in some cases a CT scan as well) as soon as possible at Great Ormond Street Hospital for Children. This involves taking a series of pictures using a special machine. We may take a biopsy using small needles under MRI guidance, for examination under a microscope. The whole process will take about 2 h. As soon as the scan is done we will arrange for your baby to be taken for the traditional post-mortem. We will ensure that at all times your baby will be treated with due respect and reverence.

The MRI scan will not delay the post mortem or the timing of burial or cremation. Taking part would not involve you in any extra hospital visits. Any additional information, if any, from an MRI/CT scan will be included in the autopsy report to the coroner. We will also need to have access to the post-mortem results and the results of any other tests that were done before or after birth. This is so that we can compare the results of tests which are done traditionally with the results from the MRI and work out which combination of tests gives the most accurate results overall.

Will my taking part in this study be kept confidential?

All information that is collected about you or your baby or during the course of the research will be kept strictly confidential. Any information we collect will only be used by the research team for the purpose of the study.

Who will have access to the case/research records?

All the data and images collected as part of this study will be stored on a secure computer. Only the researchers involved in this study will have access to the data collected in the course of this study. A representative of the hospital’s research ethics committee will also have access to the data. The 1988 Data Protection Act safeguards the use of some types of personal information. This places an obligation on those who record or use personal information, but also gives rights to people about whom information is held. If you have any questions about data protection, please contact the data protection officer via the switchboard on XXXXXX. The results from our project will be published as papers in medical journals. No data will be published that allows for individuals to be identified in any way. If requested, we will be able to send you copies of any papers published when we have completed the study in 3–4 years’ time.

Do you have to take part?

It is up to you to decide whether or not to take part. If you do decide to take part you will be given this information sheet to keep and will be asked to sign a consent form. You will be given a copy of the signed consent form for your records. If you do not feel able to take part it will not in any way affect the care your family receives.

Who do I speak to if I have further questions or worries?

In the first instance please contact Dr XXX who is coordinating this project. His contact details are given below. Drs XXXX can also be contacted if you need any further information or if Dr XXXX is not available. If you wish to speak to someone not directly involved in the study then please contact XXXXX.

If you have any complaints about the way in which the project is being or has been conducted, in the first instance please discuss them with any of the doctors listed below. If the problems are not resolved, or you wish to comment in any other way, please contact XXXXX.

Who is organising and funding the research?

This study is being organised by the Cardiothoracic, Radiology and Pathology Departments at Great Ormond Street Hospital for Children (and by the Foetal and Neonatal Medicine Units and Pathology Department at University College London Hospital). Funding is provided by the Department of Health. This study has been reviewed and approved by the Great Ormond Street Hospital Research Ethics Committee.

Thank you once again for all your time and trouble.

Contacts for further information:

XXXXXXXX

Appendix 3

Version 1.0

REC reference number: XXXXXX

Study R & D Number: XXXXXX

……….

……………….

……………….

Patient Identification Number for this trial:…………….

Consent form

Title of project: post mortem magnetic resonance imaging in the fetus, infant and child: a comparative study with conventional autopsy

Researchers: XXXXXXXXXXXXX

  1. I confirm that I have read and understand the information sheet dated xxxxx (version XX) for the above study, and have had the opportunity to ask any questions. □

  2. I understand that my participation is voluntary, and that I am free to withdraw at any time, without giving any reason, without my medical care or legal rights being affected. □

  3. I understand that sections of baby’s medical notes may be looked at by responsible individuals named in the study or by regulatory authorities from the Trusts. I give permission for these individuals to have access to baby’s records. □

I agree to take part in the above study.

Name of parent/legal guardian ________ Date ________ Signature ________

Name of person taking consent ________ Date ________ Signature ________

(if different from researcher)

Researcher ________ Date ________ Signature ________

1 for patient; 1 for researcher; 1 to be kept with hospital notes.

REFERENCES

View Abstract

Footnotes

  • Competing interests: None.

  • Funding: The work for this project is funded by the UK Department of Health Policy Research Programme (grant no. 0550004) and the British Heart Foundation (grant no. CI/05/010); neither funding body had any role in the analysis of data, results or conclusions of the study. The manuscript has been reviewed and approved by the Department of Health (UK). AMT is funded by the UK National Institute of Health Research (NIHR; grant no. SRF/08/01/018). NJR and NJS are funded by the Higher Education Funding Council for England (HEFCE). This work was undertaken at GOSH/ICH and UCLH/UCL who both received a proportion of funding from the UK Department of Health’s NIHR Biomedical Research Centres funding scheme.

  • The MaRIAS (Magnetic Resonance Imaging Autopsy Study) Collaborative Group consists of the following members: Alan Bainbridge (Medical Physics, UCL), Jocelyn Brookes (Radiology, UCLH), Lyn Chitty (Fetal Medicine, UCLH/ICH), Kling Chong (Paediatric Neuroradiology, GOSH), Andrew Cook (Cardiac Morphology, ICH/GOSH), Enrico de Vita (Medical Physics, UCL), Brian Harding (Paediatric Neuropathology, GOSH), Tom Jacques (Paediatric Neuropathology, GOSH), Rod Jones (Research MR Radiographer, ICH/GOSH), Mark Lythgoe (High field Imaging, Centre for Advanced Biomedical Imaging, UCL), Wendy Norman (Research MR Radiographer, ICH/GOSH), Oystein Oslen (Paediatric Chest and Abdomen Imaging, GOSH), Cathy Owens (Paediatric Chest and Abdomen Imaging, GOSH), Amaka Offiah (Paediatric Musculoskeletal MR, GOSH), Nikki Robertson (Neonatology, UCL/UCLH), Tony Risdon (Paediatric Forensic Pathology, GOSH), Neil Sebire (Perinatal and Paediatric Pathology, ICH/GOSH), Rosemary Scott (Perinatal Pathology, UCLH), Dawn Saunders (Paediatric Neuroradiology, GOSH), Silvia Schievano (Medical Engineering, ICH), Angie Scales (Family Liaison Sister, GOSH), Andrew Taylor (Principal Investigator, Centre for Cardiovascular Imaging, ICH/GOSH), Sudhin Thayyil (Centre for Cardiovascular Imaging, ICH/GOSH), Angie Wade (Trial Statistician, ICH). GOSH, Great Ormond Street Hospital for Children; ICH, UCL Institute of Child Health; UCL, University College London; UCLH, University College London Hospitals.

  • Ethics approval: The GOSH-ICH Research Ethics Committee approved this study.

Request Permissions

If you wish to reuse any or all of this article please use the link below which will take you to the Copyright Clearance Center’s RightsLink service. You will be able to get a quick price and instant permission to reuse the content in many different ways.