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Overcoming barriers to establishing autopsy procurement programs in pediatric patients with central nervous system tumors: a call to develop regional centers

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Abstract

Background

While autopsy-repository programs with a variety of pediatric central nervous system (CNS) tumor types are a critical resource for preclinical neuro-oncology research, few exist and there is no published guidance on how to develop one. The goal of this prospective Pediatric Brain Tumor Repository (PBTR) study was to develop such a program at Cincinnati Children’s Hospital Medical Center (CCHMC) and then publish the quantitative and experiential data as a guide to support the development of similar programs.

Methods

Protocols and infrastructure were established—to educate oncologists and families, establish eligibility, obtain consent, address pre- and post-autopsy logistics (e.g., patient and tissue transportation), process and authenticate tissue samples, and collect and analyze data.

Results

Of the 129 pediatric CNS tumor patients at CCHMC who died between 2013 and 2018, 109 were eligible for our study. Of these, 74% (81 of 109) were approached for PBTR donation, and 68% (55 of 81) consented. In the final year of the study, approach and consent rates were 93% and 85%, respectively. Median time from death to autopsy (postmortem interval, PMI) was 10 h (range, 1.5–30). In the outpatient setting, PMI increased with distance (from the hospice/home where the patient died to CCHMC). In all patients, PMI appeared to be lower, when consent was obtained more than 24 h before death.

Conclusions

Procurement of autopsy specimens need not be a barrier in neuro-oncology research. Regional centers, strict timing-of-consent, patient education, and dedicated staff are all needed to minimize PMI and, thereby, increase the value of the procured tissue for an array of basic and translational research applications.

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Acknowledgements

We acknowledge the pathology leadership of Dr. David Witte, regulatory support of Dr. Renee Doughman, psychosocial support of Mandy Bley and Dr. Aimee Thompson, coordination support by Nancy Yanez-Escorz and Daniel Heinbaugh, and the editing expertise of Dr. Rebecca Tuttle. We are grateful for the generosity of the patients and their families for supporting the study.

Funding

This work was financially supported by Sophie’s Angel Run, Inc and Joshua’s Wish Foundation

Author information

Authors and Affiliations

  1. Division of Oncology, Department of Pediatrics College of Medicine, Brain Tumor Center, Cancer and Blood Diseases Institute, Cincinnati Children’s Hospital Medical Center, University of Cincinnati, 3333 Burnet Avenue, Cincinnati, OH, 45229-3026, USA

    Mariko DeWire, Trent R. Hummel, Peter de Blank, Ralph Salloum, Natasha Pillay-Smiley, Elizabeth Gilger, Maureen Gallagher, Lori Driver, Dinah Meister, Heather Ward, Rachid Drissi, Shiva Senthil Kumar, Satarupa Sengupta, Bridget Kikta, Wanda Meriwether, Susan Jelinek, Anthony Asher & Maryam Fouladi

  2. Division of Hematology/Oncology, Department of Pediatrics, IWK Health Centre and Dalhousie University, Halifax, Nova Scotia, Canada

    Craig Erker

  3. Department of Hematology/Oncology, Dayton Children’s Hospital, Dayton, OH, USA

    Lionel M. L Chow

  4. Department of Hematology/Oncology, Phoenix Children’s Hospital, Phoenix, AZ, USA

    Lindsey Hoffman

  5. Division of Research, Department of Surgery, College of Medicine, University of Cincinnati, Cincinnati, OH, USA

    Satarupa Sengupta

  6. Division of Pathology and Laboratory Medicine, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, USA

    Christine Fuller

  7. Department of Radiology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, USA

    Blaise Jones & James Leach

  8. Department of Pathology and Laboratory Medicine, Nemours Children’s Health System, Orlando, FL, USA

    Lili Miles

  9. Center for Childhood Cancer & Blood Disorders, Nationwide Children’s Hospital, Columbus, OH and The Ohio State University College of Medicine, Columbus, OH, USA

    Rachid Drissi

  10. Center for Childhood Cancer & Blood Disorders, Nationwide Children’s Hospital, Columbus, OH, USA

    Shiva Senthil Kumar

  11. Hematology/Oncology &, BMT, Nationwide Children’s Hospital, The Ohio State University College of Medicine, ColumbusColumbus, OHOH, USA

    Maryam Fouladi

Authors
  1. Mariko DeWire
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  2. Craig Erker
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  3. Trent R. Hummel
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  9. Elizabeth Gilger
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  20. Anthony Asher
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  21. Blaise Jones
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  24. Christine Fuller
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Contributions

Conception and design: MD and MF. Data collection and assembly: AA. Laboratory: RD, SSK, and SS. Psychosocial data review: BK, WM, SJ. Data analyses and interpretation: MD and CE and pathologic analyses by LM, DW and CF. Manuscript writing and editing: MD and CE with critical feedback from all authors. Final approval of manuscript: All authors. Accountable for all aspects of the work: All authors.

Corresponding author

Correspondence to Mariko DeWire.

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The authors declare that they have no competing interests.

Ethics approval

The Institutional Review Board at CCHMC approved the protocol and continuing approval was maintained throughout the study.

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Informed consent and assent were obtained according to institutional guidelines.

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DeWire, M., Erker, C., Hummel, T.R. et al. Overcoming barriers to establishing autopsy procurement programs in pediatric patients with central nervous system tumors: a call to develop regional centers. J Neurooncol 152, 107–114 (2021). https://doi.org/10.1007/s11060-020-03679-2

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  • DOI: https://doi.org/10.1007/s11060-020-03679-2

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