The Braist Study

[mariaf]

Now for one, If my kid had scoliosis, I'd probably be willing to put my kid in a brace for a chance at saving them from surgery.
Even if I had to see the discomfort and distress on their face..
And even if there was a chance the scoliosis wasn't going to progress anyway.

But as I said before I probably would brace my kid, even if I knew there was a chance it wouldn't matter either way.
- Scott

Hi Scott,

I can't help but be curious (sorry, I didn't have time to re-read old posts to see if you shared this personal info), but do you have children? If so, I can assume from your post that you have never had to brace any of them.

I'm NOT trying to attack you - so I hope it doesn't come across that was as that is not my intent, but I honestly and TRULY believe that people may think one way, but once in that particular situation, feel quite differently. I, myself, could have said the same thing as you BEFORE I actually knew what it was like to tell a child to wear a brace 22-23 hours a day and see firsthand all of the issues that could accompany it (none of them fun).

Now, it may very well be that you would do exactly as you say/think if you were in that situation and have your child wear the brace even though they might be miserable and even though, as you say, there's a chance it wouldn't matter either way. And even if you started to see that it was affecting their entire childhood.

Or maybe you'd surprise yourself and feel differently.

Just a thought to consider.

Take care,

[jrnyc]

hi Maria
could not agree with you more...

as a kid, i would throw the night guard i was supposed to wear across
the room at nite, in my sleep, it hurt so much...
NO WAY i would have worn a brace if my scoli had been diagnosed as a kid...

i do not know how any young child can be convinced to wear a brace for the future of their spine...young kids have a hard time projecting into the future alot of the time...it seems so far away...
and to explain to them that they must suffer now for the future...
cruel, hard, tough thing to try to do...
especially when they could end up needing surgery anyway...
leading to lots of hours on a therapist's couch to deal with the
resulting anger and frustration!

jess...and Sparky

[mariaf]

Hi Jess,

As a parent, I always remind myself that they only get one childhood.

I often tell the story of what my daughter (who was going on 13 at the time) said to me when I was lamenting on whether or not surgery (VBS) was the right thing for David. I knew in my heart that it was the right choice; nonetheless, it was a heavy weight to bear, basically making the decision for him, since he was in kindergarten. She said to me "Mom, I would probably be a little afraid of surgery, but I would want to have it rather than EVER wear a brace to school". She then informed me that NO WAY could I do that to her little brother either. Come to think of it, she's been a force to be reckoned with to this day - LOL!

Jackie says "WOOF" and Happy Halloween to Sparky!

[jrnyc]

thanks...Sparky says "Boo, too"

how wonderful that your little boy has such a loving sister...
they will be such great friends...how super for her to have a
brother she is close with...
you are doing a great job, for them to be so loving and caring to
each other...

jess...and Sparky

[mariaf]

Or maybe she just likes to tell me what to do - LOL :-))

Only kidding, she definitely looks out for her little bro!!!!

[LindaRacine]

As a parent, I always remind myself that they only get one childhood.

I once heard Robert Winter, who was (and maybe still is) a bracing advocate, talk about bracing kids. He felt strongly that bracing was a good thing. He said something that has always stuck with me, and that was that he felt that while parents should encourage their kids to wear their brace, it wasn't worth ruining the parent/child relationship. I think that's really true. I've seen more than a few parent/kid relationships where the parents are very pushy about treatment (whether it's bracing, exercise, surgery or anything else). Most of those kids seem like they're very withdrawn, and I think they feel like they've had no power in terms of what is done to them.

[Pooka1]

I've seen more than a few parent/kid relationships where the parents are very pushy about treatment (whether it's bracing, exercise, surgery or anything else). Most of those kids seem like they're very withdrawn, and I think they feel like they've had no power in terms of what is done to them.

Surgery is rarely, if ever, a choice for kids in range who are healthy enough for surgery. That's my understanding. It's not a choice and not in the same category as bracing and PT, neither of which have been shown to avoid surgery for life.

It's like saying my parents were "pushy" about my having to have my diseased, about to burst, appendix removed in a surgical procedure. Courts would step in on that and I hope on the fusion surgery for most kids who would benefit. You will NEVER see a court step in on bracing even after this first BrAIST article. Or PT. Can you even imagine.

[Pooka1]

From Dr. Hey's blog (again, just for perspective)...

3) Lori [Dolan] agreed that we can't fully declare "success" with no surgery or curve less than 50 degrees at skeletal maturity, since some of those patients will continue to progress and/or have pain in adulthood and may eventually need surgery.

I liked Dr. Weinstein's comments in the blog above, but would probably be cautious about concluding that bracing prevents the need for surgery altogether, since the endpoint for their current study is only until skeletal maturity --- the game isn't over when you are done growing. In our natural history study that we are preparing for publication now, as many as 38-40% of patients with scoliosis as a teen continued to progress during adulthood. Lori did say that they are planning to do a 2 year follow-up on their cohort to see if there is any further progression. We both agreed that even long-term follow-up would be helpful -- even lifetime!! Certainly my hundreds and hundreds of adults over the years with progressive painful scoliosis would encourage us to consider such a long-term, full life approach.

In any case, it is a wonderful study and may encourage some of our guests to consider bracing, but it is a decision that requires dialogue with all parties -- especially the young lady or gentleman who needs to commit to wear the brace for at least 13 hours a day for possibly several years.

In re the last quote, notice how he is not treating bracing like perhaps chemotherapy for childhood leukemia wherein all patients get that and the vast majority beat cancer or at least 75% of childhood cancers are treatable per my understanding. Unlike chemo or radiation or whatever they do for childhood leukemias, he still says it's a "decision" requiring dialogue.

Note the distance between what surgeons like Dr. Hey and researchers like Dr. Dolan say on the one hand and what lay parents breathlessly conclude on fora on the other.

Also note that I don't think (or hope) you will find anyone who knows what they are talking about claim this study shows that a single kid has escaped surgery for life.

It is going to be so very interesting to see those final curve numbers. We might all be able to go home. Or not.

[flerc]

Emphasis added.
CHD7 Gene Polymorphisms and Familial Idiopathic Scoliosis

Tilley, Mera K. PhD*; Justice, Cristina M. PhD*; Swindle, Kandice BS†; Marosy, Beth MS‡; Wilson, Alexander F. PhD*; Miller, Nancy H. MD†
Collapse Box
Abstract

Study Design. Model-independent linkage analysis and tests of association were performed for 22 single nucleotide polymorphisms in the CHD7 gene in 244 families of European descent with familial idiopathic scoliosis (FIS).

Objective. To replicate an association between FIS and the CHD7 gene on 8q12.2 in an independent sample of families of European descent.

Summary of Background Data. The CHD7 gene on chromosome 8, responsible for the CHARGE syndrome, was previously associated with FIS in an independent study that included 52 families of European descent.

Methods. Model-independent linkage analysis and intrafamilial tests of association were performed on the degree of lateral curvature considered as a qualitative trait (with thresholds of ≥10°, ≥15°, ≥20°, and ≥30°) and as a quantitative trait (degree of lateral curvature). Results from the tests of associations from this study and the previous study were combined in a weighted meta-analysis.

Results. No significant results (P < 0.01) were found for linkage analysis or tests of association between genetic variants of the CHD7 and FIS in this study, failing to replicate the findings from the previous study. Furthermore, no significant results (P < 0.01) were found from meta-analysis of the results from the tests of association from this sample and from the previous sample.

Conclusion. No association between the 22 genotyped single nucleotide polymorphisms in the CHD7 gene and FIS within this study sample was found, failing to replicate the earlier findings. Further investigation of the CHD7 gene and its potential association to FIS may be required.

Level of Evidence: N/A
http://journals.lww.com/spinejournal...pathic.16.aspx

Do you think that parents must to believe that is reasonable to expect something similar to what happened with that first CHD07 gene study, if that last braist study would be repeated? If this is your intention in posting this, did I miss some justification yours in other post? Or I should to wait for your justification as I’m waiting the end of your demonstration about the not so much uneven distribution of the X factor explaining it (not braces) the successful in the braced group.. as I’m also waiting for the raw data example (I asked for it to you many times) showing why the successful outcome in brace group don’t talk really about the brace effectiveness as Researchers said.. sure I’m forgetting something else.

If you want that parents hates braces so much as you do, you must first to give demonstrations about why they should to think that this outcome: ‘In the analysis that included both groups, the rate of treatment success was 72 percent among children with bracing, compared with 48 percent among those under observation. The benefit increased the longer bracing was worn. More than 90 percent of the children who were successfully treated wore their braces more than 13 hours a day’ is not showing a clear brace effectiveness, as Reserachesrs, Surgeons, Organizations are saying. Instead of trusting in them, it seems they must to trust in you, but of course you must to say them who you are. They should to know if you are a known Researcher or just only someone pretending in order to convince them to not consider braces as a way to avoid surgery.

Of course is unmoral what this forum does, not forcing you to do anything about that. I'm really veryyyy far to be the only one believing it.

[LindaRacine]

Surgery is rarely, if ever, a choice for kids in range who are healthy enough for surgery. That's my understanding. It's not a choice and not in the same category as bracing and PT, neither of which have not been shown to avoid surgery for life.

It's like saying my parents were "pushy" about my having to have my diseased, about to burst, appendix removed in a surgical procedure. Courts would step in on that and I hope on the fusion surgery for most kids who would benefit. You will NEVER see a court step in on bracing even after this first BrAIST article. Or PT. Can you even imagine.

Nonetheless, it has not been unusual, even in posts on these forums, for kids to feel their parents made the surgery decision, against their will. I can't remember every story, but I think all or most of these people have had bad long-term outcomes.

I agree that if a kid absolutely needs surgery, it should be done, unfortunately regardless of how the kid feels. As I'm sure you already know, there are ways to soften the blow. Most kids are not going to go willingly into such a big surgery, so their parents have the responsibility of making an unpopular decision. I'm just saying that, sometimes, I've met people who blame their parents for making them have surgery, and who do not have a good relationship with their parents (either because of the decision, or because it was the decision that broke the camel's back).

--Linda

[flerc]

They should to know if you are a known Researcher or just only someone pretending in order to convince them to not consider braces as a way to avoid surgery.

Of course I was just only referring to parents without enough science background .Certainly this not ending work done here, can confuse them very much.. they are really defenseless.

[Pooka1]

Do you think that parents must to believe that is reasonable to expect something similar to what happened with that first CHD07 gene study, if that last braist study would be repeated? If this is your intention in posting this, did I miss some justification yours in other post? Or I should to wait for your justification as I’m waiting the end of your demonstration about the not so much uneven distribution of the X factor explaining it (not braces) the successful in the braced group.. as I’m also waiting for the raw data example (I asked for it to you many times) showing why the successful outcome in brace group don’t talk really about the brace effectiveness as Researchers said.. sure I’m forgetting something else.

If you want that parents hates braces so much as you do, you must first to give demonstrations about why they should to think that this outcome: ‘In the analysis that included both groups, the rate of treatment success was 72 percent among children with bracing, compared with 48 percent among those under observation. The benefit increased the longer bracing was worn. More than 90 percent of the children who were successfully treated wore their braces more than 13 hours a day’ is not showing a clear brace effectiveness, as Reserachesrs, Surgeons, Organizations are saying. Instead of trusting in them, it seems they must to trust in you, but of course you must to say them who you are. They should to know if you are a known Researcher or just only someone pretending in order to convince them to not consider braces as a way to avoid surgery.

Of course is unmoral what this forum does, not forcing you to do anything about that. I'm really veryyyy far to be the only one believing it.

How certain are you that you are interpreting these results correctly and not over-stating things?

From the Editorial in the NEJM that was published along with the first pub from the BrAIST study written by Eugene J. Carragee, M.D. and Ronald A. Lehman, Jr., M.D... (emphasis added)

September 19, 2013DOI: 10.1056/NEJMe1310746

Although the authors initially intended to conduct a strict randomized trial, in practice this was not possible. Strong treatment preferences limited enrollment in the randomized trial, so the investigators added observational patient-preference groups. Of 242 patients included in the analysis, 116 had received randomly assigned care and 126 received patient-directed care (71% of the patients in this group choosing brace treatment). Furthermore, very few patients wore the brace for the recommended 18 hours per day; 27% of the patients stopped using the brace completely (see the Supplementary Appendix of the article, available at NEJM.org).

The difficulties with enrollment and compliance must be considered in interpreting the results. Because much of the data were derived from a nonrandomized cohort, the magnitude of the associations between brace wear (and its duration) and good outcome may have been inadvertently magnified if patients whose curves were more likely to progress were correspondingly less inclined to wear a brace (e.g., if they had relatively stiff curves, which resist the corrective pressure of the brace, or rapidly progressing curves, which outgrow the mechanics of the brace). The intention-to-treat analysis showed the same general trend of treatment effectiveness but with much less statistical confidence.

The decision to commit a 12- or 13-year-old patient to several years of brace wear requires careful consideration of both the benefits and the downsides. Although brace wear in adolescent idiopathic scoliosis does not have the serious physiological side effects that are apparent in poliomyelitis-related scoliosis, it carries financial, emotional, and social burdens that need to be considered. Overall, the findings of the present trial confirm the general proposition that brace treatment confers benefits for some patients with adolescent idiopathic scoliosis, but an open question is the applicability of the findings to individual adolescents with idiopathic scoliosis. Patients with adolescent idiopathic scoliosis represent a heterogeneous group, and this study does not have adequate numbers to evaluate the treatment effect for specific structural types of adolescent idiopathic scoliosis or the efficacy according to relative correct-ability or relative discomfort of bracing that requires high forces to maintain correction. With improved flexibility, especially in the lumbar spine, the probability of success with bracing is likely to be greater.

As the authors appropriately point out, 48% of the untreated patients had a successful outcome, as did 41% of the patients in the bracing group who spent little time wearing the brace. In retrospect, the bracing indications described are probably too broad, resulting in what may be unnecessary treatment for many patients. We agree with the authors that the equally important finding of this study is that so many growing children with adolescent idiopathic scoliosis seem to do just fine with no treatment at all; the challenge for the field going forward is to identify children who are most likely to benefit from bracing and those who are unlikely to benefit.8,9

By the way, I earlier mis-stated that 75 kids were braced in the intent to treat arm. I was wrong... only 51 were braced in that arm. That means the quartiles in the dose-response curve each contain 51/4 = 12.75 kids. So 25.5 kids wore the brace for >12.9 hours (total in the two top quartiles). That is why the comments about the groups not being large enough for a heterogeneous condition. And we still don't know what their curve magnitudes were after the bracing. If they were north of 40* then we can go home on the dose-response curve in my opinion.

The other thing I wanted to say on this issue of not having enough patients is the study was stopped early before they could recruit enough patients. I don't think it's fair to gig the researchers on not having enough patients if you are going to stop their study. What am I missing? There are not enough patients but it is no fault of the researchers. It's almost like the panel didn't want a definitive answer and made sure the number of patients would be too low to settle the question.

I would have thought that once they showed that the two groups (intent-to-treat and as-treated) were similar enough they could just combine them. But apparently not. That is why the dose-response curve is ONLY for intent to treat and therefore has too few patients.

[Pooka1]

Of course I was just only referring to parents without enough science background .Certainly this not ending work done here, can confuse them very much.. they are really defenseless.

Here are some more comments from a surgeon (Dr. William Taylor... http://neurosurgery.ucsd.edu/william-taylor-md/) and more from Dr. Carragee... (emphasis added)

http://www.medpagetoday.com/Orthoped...hopedics/41732

While the study adds the "weight of the literature" in favor of bracing, Taylor noted that the patients might have been self-selected to have success, since about 65% of those screened and eligible did not take part in the study.

Indeed, the enrolment difficulties "must be considered in interpreting the results," commented Eugene Carragee, MD, of Stanford University School of Medicine in Stanford, Calif., and Ronald Lehman, MD, of the Walter Reed National Military Medical Center in Bethesda, Md.

In an accompanying editorial, they said that the associations between braces and treatment success might have been "inadvertently magnified" if participants whose curves were more likely to progress were less inclined to wear a brace -- something might have happened, for instance, if a patient had a relatively stiff curve that resisted the pressure of the brace.

Since the study also showed that some children did well without a brace, they concluded that "the challenge for the field going forward is to identify children who are most likely to benefit from bracing and those who are unlikely to benefit."

[Pooka1]

You know, instead of averaging within quartiles, they COULD HAVE ALSO plotted all the 51 data points on the dose-response curve. Had they done that it would have been obvious that kids who wore the brace for many hours still were not <50* and that kids who wore the brace fewer hours were still <50* at maturity. The scatter would have been a take home message also.

And by the way, those 90%-93% of kids in the top quartile constitutes just under 12 kids. Call it 12. TWELVE KIDS. Once we know the curve magnitude of those 12, I don't think it will be too early to know if we can all go home.

[hdugger]

By the way, I earlier mis-stated that 75 kids were braced in the intent to treat arm. I was wrong... only 51 were braced in that arm. That means the quartiles in the dose-response curve each contain 51/4 = 12.75 kids. So 25.5 kids wore the brace for >12.9 hours (total in the two top quartiles).

...
And by the way, those 90%-93% of kids in the top quartile constitutes just under 12 kids. Call it 12. TWELVE KIDS. Once we know the curve magnitude of those 12, I don't think it will be too early to know if we can all go home.

Jumping in for a few quick corrections,

Based on the discussion of Figure 2 in the report:

* 116 braced children were measured in the dose-response curve and I do not see any note of how they're broken down between randomized and unrandomized.
* The quartiles are divided by hours of wear, and not by numbers of children. You have no way of determining the number of children in each of the quartiles.
* The top two quartiles (not just the top one) are associated with roughly 90% success.
* The dose response was deemed significant "The quartile of duration of brace wear was positively associated with the rate of success (P<0.001)." Significance testing takes the number of subjects into account - whatever the number in each group, there were enough to make the dose response results significant.

The other thing I wanted to say on this issue of not having enough patients is the study was stopped early before they could recruit enough patients. I don't think it's fair to gig the researchers on not having enough patients if you are going to stop their study. What am I missing? There are not enough patients but it is no fault of the researchers. It's almost like the panel didn't want a definitive answer and made sure the number of patients would be too low to settle the question.

Hold on a second while I turn the world right side up. This, from the paper - "The trial was stopped early owing to the efficacy of bracing." So, more or less the exact opposite of what you state here. I believe the board called the study at either the point at which bracing was deemed effective enough that it would no longer be ethical to assign children to the non-bracing group, or at the point at which there was no reason to go on with the study because they had proved their conclusion. In either case, it was called due to proven effectiveness and not the reverse.

OK, that's enough for this weekend. There's lots more that could be corrected, but that's all I have time for.