Retraining and control therapy for pediatric psychogenic non-epileptic seizures (+ 1 year follow-up), Fobian et al, 2020

cassava7

cassava7

Senior Member (Voting Rights)
Objective

Our aim was to conduct a pilot randomized controlled trial of a novel cognitive behaviorally based intervention for pediatric PNES called Retraining and Control Therapy (ReACT).

Methods

Participants were randomized to receive either eight sessions of ReACT or supportive therapy, and participants completed follow-up visits at 7- and 60-days posttreatment. The primary outcome measure was PNES frequency at 7-days posttreatment. Eligibility criteria included children with video-EEG confirmed PNES and participant/parent or guardian willingness to participate in treatment. Exclusion criteria included substance use, psychosis, and severe intellectual disability. Forty-two patients were assessed for eligibility and 32 were randomized. ReACT aimed to retrain classically conditioned, involuntary PNES by targeting catastrophic symptom expectations and a low sense of control over symptoms using principles of habit reversal. Supportive therapy was based on the assumption that relief from stress or problems can be achieved by discussion with a therapist.

Results

Twenty-nine participants (Mage = 15.1 years, SDage = 2.5; 72.2% female; 57.1% Caucasian, 28.6% African American) completed 7-days postprocedures. For PNES frequency, the Wilcoxon Rank Sum test statistic was 273.5 yielding a normal approximation of Z = 4.725 (P < 0.0001), indicating a significant improvement in PNES frequency for ReACT at 7-days posttreatment compared to supportive therapy. Participants with PNES in the 7-days posttreatment were removed from the study for additional treatment, resulting in no 60-day follow-up data for supportive therapy.

Interpretation

ReACT resulted in significantly greater PNES reduction than supportive therapy, with 100% of patients experiencing no PNES in 7 days after ReACT. Additionally, 82% remained PNES-free for 60 days after ReACT.

https://onlinelibrary.wiley.com/doi/10.1002/acn3.51138
 
One-year follow-up of treatment outcomes and patient opinions of Retraining and Control Therapy (ReACT) for pediatric functional seizures


Highlights

• Retraining and Control Therapy (ReACT) improves FS compared to supportive therapy.

• This study demonstrates maintenance of FS treatment outcomes for at least one year.

• Children with FS and their parents believe ReACT is helpful for FS.

• Targeting mood is not required to treat or prevent relapse of child FS.

Abstract

Retraining and Control Therapy (ReACT), a short-term treatment for pediatric functional seizures (FS), has been demonstrated to improve FS in children compared to supportive therapy. However, long-term maintenance of FS-reduction after ReACT is unclear.

This study aims to assess seizure frequency 1 year after ReACT and determine patient and parents’ opinions of ReACT. Children with functional seizures who previously completed ReACT and their parents were asked to report 30-day FS frequency 1 year after completing ReACT. They also reported if ReACT was helpful.

Paired samples t-tests were used to compare FS frequency before ReACT to 1 year after and to compare FS frequency in 30 days after ReACT to 1 year after.

Fourteen children (Meanage = 15.43) and their parents participated. Seven-day FS frequency for patients at 1-year follow-up (Mean = 0.15) was significantly lower than 7-day FS frequency pre-ReACT (Mean = 5.62; p = 0.005).

No differences were found when comparing FS frequency during 30 days post-ReACT (Mean = 0.29) and in 30 days before 1-year follow-up (Mean = 0.71).

This study confirms long-term maintenance of FS-reduction after ReACT and supports the efficacy of targeting FS directly as opposed to mood or stress for reducing FS. Additionally, children and parents believe ReACT is beneficial.

https://www.sciencedirect.com/science/article/pii/S2589986421000770
 
Therapy design

This trial is interesting in that although the therapy was CBT-based, it did not try to address possible underlying psychopathology — depression, anxiety or trauma from adverse life experiences — as in the CODES trial (for adults) but instead focused on trying to maintain control over PNES symptoms.

Where CBT in CODES is “a Beckian approach predicated on the assumption that for patients with DS life experiences and trauma result in maladaptive core beliefs (negative schemas), leading to cognitive distortions and somatic symptoms”, the ReACT therapy is instead “aimed at retraining classically conditioned, involuntary PNES by targeting catastrophic symptom expectations and a low sense of control over symptoms”.

Participants

Participants in this trial were teenagers of on average 15 years old and were assigned either to the ReACT therapy (n = 17) or supportive therapy (n = 15) arms.

Results

Unfortunately, the 1 year follow-up data is uninterpretable. It includes 14 out of 17 patients who were assigned to the ReACT therapy but not the patients in the control arm, because the latter received the therapy after the end of treatment as they continued to have seizures. Thus, the only available reliable data are that of the trial.

The intervention seemed quite effective as the trial was stopped early despite the small number of participants. However, there were 3 participants lost to all follow-up, all of which were in the control arm (of 15 participants, initially), and their data were not imputed. This represents 20% of the control arm. It is not known if they stopped because they stopped having seizures (which would have weakened then results of the trial) or if they found the intervention wasn’t effective (strengthening the results), or for another reason.

Of note is that the participants who received supportive therapy had a weekly mean of 3 seizures with a standard deviation of 2.5 at the 7-day post-treatment outcome (i.e. a lower bound of 1 seizure per week), so it is possible that some of them could have improved and completely stopped having seizures by the 60-day post-treatment outcome — but, as noted above, they all received therapy so we do not know if that could have happened —. The only interpretable outcome is thus that at 7-day post-treatment.

Although the intervention arm had a mean of 9.9 seizures per week with significant variance (standard deviation 12.5), it is remarkable that the participants:

- achieved no seizures at all in the week following the end of treatment and maintained only 0.56 +/- 1.37 mean seizures per week at 60-day post-treatment

- achieved a mean improvement of 14 points on the pediatric quality of life scale (Peds-QL, child self-report) in the week following treatment, compared to 2.3 points in the control arm

- needed only a mean of 4.6 +/- 1.9 sessions before attaining remission even though 8 sessions were planned in total.

Questions and concerns

My concern is that in the intervention arm, sessions 2 and 3 introduced “reinforcers” to reward progress and sessions 4 and 5 “punishers”, which may have incentivized children to underreport seizures that occurred in the absence of their parents or to hide them in their presence. The follow-up paper reports that out of 13/17 participants, only 2 (15.4%) found that parent involvement was beneficial. The paper also does not detail how differing occurrences of seizures were handled: “discrepancies in PNES reported between the participant and parent were discussed in person, and a consensus was reached”.

Another concern is that on the Adolescent Coping Orientation for Problem Experiences (ACOPE) scale, the paper interprets positively statistically significant between-group reductions in coping behaviours that seem healthy in teenagers, such as relaxing, being humorous and self-reliance/optimism. There was, however, a reduction on the “avoiding solving problems” item.

Further, there was no within-group change in the intervention arm with regards to anxiety and depression, which seems odd given the reported remission of their seizures and subsequently improved quality of life.

Perhaps the oddest point is that at follow-up, only 6/13 respondents (46.2%) found it helpful to ”have a plan to address episode symptoms and [to learn] to control episodes” while this is the very nature of the therapy. Further, 5/13 (38.5%) found it helpful to “have someone to talk to”, so they might have benefited from supportive therapy alone. This casts doubt over the purported mechanism of action of the ReACT therapy.

Finally, 86% of them were seizure-free during the month following treatment, but only 57% were during the month before the 1-year follow-up assessment. This suggests a loss of effectiveness over time, especially given the 4 participants who were lost to follow-up. The number of weekly seizures seems to have remained reduced post-treatment and at follow-up, though (mean 0.29 vs 0.71).

Conclusion

Overall, the lack of follow-up data makes it hard to tell whether the intervention was truly effective, though it seems to have been. This pilot study probably warrants a larger replication.

Edit: the second author of the follow-up paper, Jerzy P. Szaflarski, is the associate editor of the journal in which it was published, Epilepsy & Behavior Reports.
 
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This quote from the first paper is interesting.
Despite the significant prevalence, there are no published randomized clinical trials (RCTs) assessing the treatment of pediatric PNES. However, there have been a few RCTs published assessing treatment of adults with PNES. Based on these studies, psychotherapy appears to be a promising treatment in pilot studies. However, only between 25% and 56% of patients have achieved complete PNES remission in these studies,20-22 suggesting that further refinement of psychotherapy-based interventions is needed. These treatments use the psychoanalytic explanation for FND and target fear avoidance or work to attribute patients’ symptoms to psychosocial issues.22, 23 In the largest RCT assessing CBT for PNES in adults by targeting fear avoidance, there was no difference in PNES between CBT with standard medical care and standard medical care alone at 12 months after treatment.24 These negative results suggest mood is not the most effective PNES treatment target and support the investigation of novel treatment targets.
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My first observation is that treatment is routinely being provided to young people with PNES in the absence of evidence. This is not evidence-based medicine.
The second is that the evidence for psychotherapy being useful in adult PNES patients is extremely weak to the point of suggesting that it does not help. Still not evidence-based medicine.

The authors of these two papers sought to offer a 'new! improved!' type of psychotherapy. This is based on the observation that some PNES patients don't seem to have emotional trauma. They have settled on the flaw in the person being that they don't think they can control their seizures and that they catastrophise.
The etiological understanding of PNES has evolved over time. Providers often explain PNES as a physical manifestation of psychological distress from disturbances in personal relationships, stress, childhood abuse, or trauma.9, 10 However, some patients with FND do not have a comorbid psychiatric diagnosis or trauma history, and for the ones who do, it is unclear if (and if yes, how) it is related to the etiology of their FND.11, 12 This suggests trauma and/or psychopathology may not be the most effective targets for PNES intervention.

Recent research has provided evidence for two additional risk factors associated with PNES: catastrophic symptom expectations and perceived control over symptoms. Children with PNES have greater catastrophic symptoms expectations such as, “When my head is pounding, I worry I could have a stroke,”13, 14 which can result in the occurrence of the expected symptoms.15 Additionally, children report no control over their PNES,16 indicating that children perceive PNES to be involuntary. Experimental research in adults has confirmed the finding of impaired perceived control over actions in patients with FNDs.17-19This supports the development of a novel PNES intervention targeting these factors.
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Surely it is reasonable for a child whose head regularly pounds and experiences seizures to feel more concerned about their symptoms than a child who does not experience these or any other serious symptoms? Surely the child who does not feel some concern about having seizures is the one who has the abnormal reaction?

(edit to fix grammar)
 
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Forty-two patients were assessed for eligibility and 32 were randomized.
3 were lost to follow-up, all in the support therapy arm, leaving 17 in the REACT treatment arm and 12 in the support therapy arm.

As shown in Figure 1, 71% of the ReACT group and 67% of the supportive therapy group completed all eight therapy sessions. Participants’ reasons for ending treatment early differed by therapy assignment, with those in ReACT discontinuing because of PNES cessation and those in supportive therapy discontinuing because of worsening of or no improvement in symptoms or new cancer diagnosis.
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I can't help but wonder if the cancer caused the seizures; it can happen. 2 patients in the REACT arm and 1 patient in the supportive therapy arm also had diagnoses of epilepsy (I find the idea that someone can have both real epilepsy and PNES a bit ridiculous. If someone with epilepsy can sometimes have seizures that don't show the right brainwaves, then presumably someone diagnosed with PNES might have epilepsy as well - it's just that the day they were assessed, their seizure didn't meet the criteria for epilepsy).

cassava7 said:
My concern is that in the intervention arm, sessions 2 and 3 introduced “reinforcers” to reward progress and sessions 4 and 5 “punishers”, which may have incentivized children to underreport seizures that occurred in the absence of their parents or to hide them in their presence.
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Yes, that's a really good observation. There is reference to rewards and punishers in both therapy descriptions, but they could have been applied differently in each therapy. It is likely that the REACT therapy, with its suggestion that thinking differently will cure the seizures would be a lot more likely to induce false reporting of improvement than the supportive therapy where the therapist just made empathetic noises as the child talked about their stressors.
The provision of supportive therapy as a standard of care is based on the assumption that relief from stress or problems can be achieved by discussion with a therapist. The purpose is not to acquire new skills or find solutions to problems.
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