It was back in the summer of 2005 that I first became aware of any institution making a concerted effort to obtain DIPG samples for research purposes. On a visit to Memphis, I found out that researchers there were going to attempting to put a comprehensive IRB proposal (and place in clinicaltrials.gov) together to formalize a process for post-mortem DIPG donation. At that time, France had just started the biopsy trial and to me this was an American attempt to push DIPG research also. To me it was an exciting time- the first time I had heard of anyone really trying in an organized way to look at DIPG genomics in the US.
Since it was something that was not done, there were concerns on how to approach families- and if they would even donate. To try to answer some of these issues, specific sections of the research would examine parental feeling towards autopsy through a decisional regret survey and a 7 question survey. Questions included items such as reasons for participating in the study, what was good and bad about participating and do you have suggestions. In February an article came out electronically ahead of print in the Journal of Pediatrics detailing the parent experience.
Thirty three parents of 32 children answered the the survey (82% participation). Of these 18 received care at St Jude and 14 at other institutions. Those that received care at other institutions were in contact with St Jude directly- some specifically because of awareness of the autopsy program. Parents completed the in a mean time of 11 months.
None of the parents expressed regret in participation. The parents indicated that they consented to autopsy to help other families in the future and to help other parents know that they are not alone.
Parents also said that:
* it was better if the primary physician asked- specifically someone who "they had a relationship with and who showed concern."
*there is "no right time" to initiate this discussion. Parents do recognize that this is tough for the docs as well. Most said that they would have preferred an earlier discussion. Some said that an earlier discussion was less comforting. Timing of the discussion will likely take an individual approach. (And from a personal point of view- a mother and a father might be at different places when considering this discussion.)
* there was a need to have clear information about procedures. Knowing specifics helped decrease anxiety. Some of these specifics included exactly what would be done during autopsy and how the child would look afterwards.
One of the specific issues addressed early was funding of transportation and autopsy. Since many of children treated at St Jude die at home and not close to the facility, the logistics of funding had to be considered before this research could even be started. It was at this point that a DIPG family was looking to make a difference. Tyler's Treehouse agreed to fund non-covered expenses associated with post-mortem donation in order to make this research happen.
Tyler's Treehouse is named for a 5 year old boy- the 3rd of 4 sons. Tyler was diagnosed on January 30, 2006 having symptoms only for 1 week. The family went to St Jude but Tyler's tumor was too advanced to even start treatment. He died a week later on February 8, 2006.
Without a foundation support like Tyler's Treehouse this type of program may not have been able to get started. Thank you to the Scott's for their actions in their time of grief.
The logistics of making a DIPG Post Mortem Donation Program are not easy- but it is possible. And from such programs we are beginning to understand DIPG. Research is possible from autopsy specimens. The effort has made a difference.
References:
DNA Analysis of Tumor Tissue Samples from Patients with Diffuse Brain Stem Glioma (NBTP02)
http://clinicaltrials.gov/ct2/show/NCT00899834?term=St+Jude+brainstem+glioma&rank=2
Bereaved Parents' Intention and Suggestions about Research Autopsies in Children with Lethal Brain Tumors
J Pediatr. 2013 Feb 19. pii: S0022-3476(13)00039-5. doi: 10.1016/j.jpeds.2013.01.015. [Epub ahead of print]
http://www.ncbi.nlm.nih.gov/pubmed/23433673
Somatic Histone H3 alterations in pediatric diffuse intrinsic pontine glioma and non-brainstem gliobastomas
http://www.ncbi.nlm.nih.gov/pubmed/22286216
