How do you pick which clinical trials to join?

In Q&A during ANR2016, prof Glenn Marshall asked me “How do you pick a clinical trial?”.

This is an incredibly hard and loaded question to answer, but I wanted to give a shot answering it within my context, and hopefully hear some more from others on this topic.

Where we are today?

Shalev was diagnosed in October 2015 with MYCN amplified stage 3 Neuroblastoma. We have been treated under SIOPEN high risk protocol and been through rapid Cojec induction, Surgery, BuMel transplant, local radiation and have just started immunotherapy (first round of CH 14:18/CHO in 2 weeks).

The tumour has responded very well to chemotherapy and was completely removed during surgery. We are lucky to be in one of the “best case scenarios” for HRNB.

Our immunotherapy will not include IL2 or GM-CSF as SIOPEN have recently dropped it from the study.

If we are lucky and continue with this trend, we are looking at finishing our frontline treatment in December 2016.

If all goes according to plan there are 2 numbers we are faced with.

  • There is a 25%-35% chance of relapse after we are done treatment if all goes well.
  • The first 6 months after treatment ends are the riskiest, historically half the relapses will happen in the first 6 months.

How would I go about picking a trial?

As a parent even 1% chance of relapse is a hard number to live with. We want our kids to be cured and for all to be done.

It is incredibly hard living with the odds around HRNB and going back to normal life.

I am torn between wanting to keep on going with treatment till I know there is only a tiny chance of relapse, to commencing normal life.

One tragedy with Neuroblastoma is that some are receiving highly aggressive treatment that could end up causing other disease despite being cured. There is a chance we were done with our Neuroblastoma after surgery and that it would never come back (say 5-10%) but there was an even more significant chance it would come back. So we keep treating, until our odds are better. Despite possibly being cured many months ago.

When evaluating trials I have a few sources:

  • My oncologist and team are my primary source of advice. As it stands there is no clinical trials available in Sydney after frontline therapy.

  • The internet, with all its curses, is another source of advice. In particular I can read through https://clinicaltrials.gov/ searching for Neuroblastoma. I can ask advice from other parents (internationally as well) online.

  • I can seek advice from other oncologists and specialists in the field

Should we consider any phase 1 trials?

The first question that is trivial to answer is “Should I consider any phase 1 clinical trials?”

Phase 1: Studies that are usually conducted with healthy volunteers and that emphasize safety. The goal is to find out what the drug’s most frequent and serious adverse events are and, often, how the drug is metabolized and excreted.

Firstly, there are no existing Phase 1 trials available for healthy patients that just completed HRNB protocols so the question is kind of moot.

Even if there was an available Phase 1 trial I would not be interested as

  • The goal is to determine toxicity, I would be risking making my child very ill for a complete unknown.

  • What about long term effects?

I would definitely consider phase 1 trials if I was at the end of my tether, and looking at a very morbid prognosis. But given stuff is reasonably positive, I see zero reason for layering any more risk.

Should we consider phase 2 trials?

This is where stuff gets a lot more tricky.

As it stands there are 2 trials I have been investigating that are available after frontline treatment:

(There is a 3F8 trial that also seems to be available, though it does not make sense to go through the pain of immunotherapy again after already being through it with CH 14:18)

The goal of phase 2 trials is:

Phase 2: Studies that gather preliminary data on effectiveness (whether the drug works in people who have a certain disease or condition). For example, participants receiving the drug may be compared to similar participants receiving a different treatment, usually an inactive substance, called a placebo, or a different drug. Safety continues to be evaluated, and short-term adverse events are studied.

Meaning, we are not sure if the treatment will help or not, but at least we are trying to figure out.

The enormous cost of overseas treatment

Sometimes, as parents, we like to think that any cost to increase odds is reasonable. Yes, I will mortgage my house to increase the odds my son will be cured.

The reality, of course, is more complicated:

  • The monetary cost of even a trivial trial in the US can be astronomical.

For example: take the DFMO trials. The drug itself, Elfornithine, is listed under the WHO Model List of Essential Medicines. It is easily accessible in all countries and been used for many years for sleeping sickness and facial hirsutism.

On the face of it the cost for such a trial should be low. But… if you live in Australia or Europe or anywhere that is not the USA the cost skyrockets.

  • To participate in the trial you must travel to the US every 3 months (estimated cost $6000 AUD * 8 = 48,000AUD =~ 35K USD)

  • To participate in the trial you must complete scans in the participating hospital every 3 months, this will cost approx 10K USD per scan which add up to 80K USD.

So we are already up to 115K USD just for the privilege of helping figure out if DFMO actually helps.

Similarly the cost of completing the vaccine trial can be enormous as well, though I am finding it much harder to estimate. It will include at least 4 comprehensive scans and more 6 or so trips to the US including first 1-2 months where it makes no sense to complete a 20 hour journey back to Sydney just to return in a week or 4.

Add on to this, there are a pile of other risks and costs that can not be ignored

  • The risk of getting sick during frequent air travel

  • The risk of the negative effect on siblings during this period (especially if relocating temporarily)

  • The strain on the family during this time

The unknown nature of phase 2 trials

A medication or treatment is still in an unknown state during phase 2

  • It may do nothing
  • It may cure
  • It may cause harm (short term or long term)

At the end of the day, only time we “know” a medication is sure to help is after it is out of phase 2 (and ideally phase 3 trials). During phase 2 we do not know. We like to think that preliminary results are “promising” or that “mouse models” showed incredible promise but we just don’t know.

We do not know if the vaccine helps or not, we guess that odds are it does not cause any long term harm, historically no latent effects have been pinned on the immunotherapy drugs. But… we just don’t know. Which is why we are running the trial.

Similarly we do not know if the DFMO trial helps. We hope it does, but we do not know.

When should I start a preventative trial?

One key piece of information our NB trends teach us is that if we are going to be improving relapse rates we got to start treating back-to-back after the frontline treatment.

For example, if we are to survive without relapse for 6 more months after we finish treatment our odds of event free survival jump up to 82-87%, our odds for event free survival for the 2 following years become around 91%.

It is my view that any trials looking at prevention should strictly start within days of finishing frontline treatment. Otherwise we miss out on the most risky period. There is going to be some significant statistical variation between people who start a trial a day after frontline therapy and 2 months after it.

The impossible task of interpreting initial results

In the case of the DFMO trial we have recently seen initial results 2 year EFS for 91% (± 4%). Which on the face seem much better that the 50% event free survival we get when we started treatment.

But … it’s complicated:

  • The longer we survive, the better our odds are
  • The less severe side effect we have experienced the better our odds are (what about the children with permanent liver of kidney damage that are excluded from trials)
  • If we happen to have survived event free 6 months after frontline we are expecting 2 year EFS of 90%+

We can not cherry pick numbers that feel good. If we want to make informed decisions we have look at published, peer reviewed publications and have to consult experts.

Where do I stand today on the 2 phase preventative trials?

If I was living in New York, I would certainly consider the vaccine trials. However, it is super hard to justify the enormous cost of travel and uprooting family just to get access to the trial.

As a parent I am angry that the vaccine trial is only available in NY, I feel that science could progress significantly faster if MSKCC made the vaccine available elsewhere and we could answer the question of efficacy significantly faster.

With the DFMO trial there are still no published results, and I find it incredibly hard to justify the international travel and huge cost. If I lived closed to a participating center I would consider it provided the concerns raised by COG and SIOPEN are addressed.

Navigating the maze of even 2 trials is an enormously complicated experience. I don’t envy the pain many of the relapse families are going through when they have no option but to pick 1 of 100s of open trials.

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Great summary - I watched the videos from ANR and if that was you asking all the questions on the left of the room fair play to you!.

I think parents everywhere are torn over dfmo - here it is a major talking point as it has been in the media a lot after some high profile fundraising campaigns to the extent where people who know nothing about Neuroblastoma ask you if you are going to go to Michigan for the cure!

Like Australia it is a huge financial and logistical undertaking to go for dfmo without having seen solid results debated by peers. Julie park in her talk made a very interesting analysis. They made me think why (if we are lucky enough to survive frontline therapy and go NED) would I want to continue to make Neuroblastoma part of my child’s life for two more years by enrolling her in dfmo an ocean away.

She could be one of the lucky ones and never relapse then I would have taken two healthy years for our family and made them focus on cancer as well as the massive financial impacts. alternatively she may relapse after DFMO and then I would be financially exhausted and unable to get treatment for relapse. I have two other kids and have to consider the impact on them also.

Sure if I lived in Michigan it would be a different scenario but then I would consider also do I want to do another approx 8 neuclear scans on a child in remission and while dfmo has no significant side and the scans are relatively safe I would be concerned about cumulative effects on her young body.

It’s a minefield and I’m not ready to make any decisions yet.

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Here are two recent sets of data released on dfmo -

http://aspho.planion.com/Web.User/AbstractDet?ACCOUNT=ASPHO&ABSID=12199&CONF=AM16&ssoOverride=OFF&CKEY

http://anr-2016.p.asnevents.com.au/days/2016-06-22/abstract/33202

The population of patients treated reported in the first link is 90 but in the second link it is 94 of which only 91 were considered in the analysis. Also the sub population from the antibody trial is reported as 73 in one and 74 in the other. We’ll likely have to wait to formal publication of the trial results in papers to see why these discrepancies exist. Possibly due to data handling approaches. But these are unusual in my mind.

Hi Sam @sam ,

Do you know of where I can get the information about these concerns? I saw the video from the latest NB conference the Dr. Julie Park gave her opinion about the treatment.

Do you know about some other sources?..

Thanks!

The COG concerns are mostly in Julie’s talk, the SIOPEN one’s are the same mentioned by my Oncologist. David Zeigler is in charge of DFMO relapse trial that are running in Sydney.

Perhaps have a chat with your team about DFMO and get some feedback from them as well?

Yes, this is my plan. I will post their comments here…

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We discussed it and our onc is very skeptical. Concerns on dosage, validity of data, overall difference to cog standard outcome etc.

Well, I for one can assure the data is valid.

Kyle to you know the reason for the differences in reported numbers in the links above?

Yes, the abstracts were submitted prior to the study being completed (Dec) and each were due on different dates. The most updated data was submitted at each due date.

The study completed Feb 2016. The data presented at the conference was the most updated data - the number of patients is now final. The data will continue to mature as time goes on.

There seems to have been some recent relapses on the dfmo trial given that these kids were enrolled before the close of the trial in Feb when will the data be readjusted to account for them. An informal number of recent relapses based on the posts in web pages by parents is 2 more possibly 3. Based on a patient population of 94 this represents another approximately 3 % relapse rates - granted this is an estimated number but the facts reported by parents are that kids on dfmo and finished the dfmo trial are unfortunately relapsing. Can anyone give the latest official figures of number of patients enrolled and number of relapses ? Will there be a 3 yr review?

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Are all of them for the first remission or after the first relapse? I saw one post with such recent story…

Over the past week I have seen 3 parents post that their kids relapsed in the trial and as far as I know you must not have relapsed before the trial so by default all are 1st relapse

If I am not mistaking this is the original (closed now) trial:
https://clinicaltrials.gov/ct2/show/NCT01586260

Inclusion Criteria:

  • Age: 0-21 years at the time of diagnosis.
  • Diagnosis: histologic verification at either the time of original diagnosis or a previous relapse of high risk neuroblastoma.

For me it looks like it is open for everybody…

Also there is an old post from this forum where there is a story about taking DFMO after relapse:

But I would agree with you that there is a lack of official updates from that trial. Especially, when you suppose to pay huge amount just to participate in this trial…

Quick follow up question - the clinical trials.gov website updated may 10th says enrollment complete at 89 patients this is after the abstracts. So is this the final number?