Acceptance and commitment therapy in youth with neurofibromatosis type 1 (NF1) and chronic pain and their parents: A pilot study of feasibility and preliminary efficacy

Probably not a substantial claim: may not pass S3 (load-bearing).

A growing body of literature indicates that even brief mindfulness-based interventions can reduce reported acute and chronic pain.

“A growing body of literature has found that even brief mindfulness-based interventions can reduce reported levels of acute [Zeidan et al., 2012] and chronic pain [Ussher et al., 2014].”

• cites Zeidan et al., 2012 sourceSupported
• cites Ussher et al., 2014Not matched

  Couldn’t be matched to a paper in Crossref, so it can’t be checked.

May combine two claims.

NF1 differs from other chronic pain conditions in that its pain is probably neuropathic and many patients have learning and attention problems.

“However, NF1 is distinct from these other conditions in terms of the probable neuropathic nature of the pain [Ferner et al., 2004] as well as the fact that many individuals with NF1 have learning and attention problems; thus, investigations on the feasibility and efficacy of ACT within this population are needed.”

• cites Ferner et al., 2004 sourcePartly supported

  The citation is attached to the proposed neuropathic nature of NF1 pain, not the statement about learning and attention problems. The Discussion (page unavailable) reports neuropathic pain in one patient, but says neurofibromatous neuropathy was painless in seven of eight patients; the paper does not establish that NF1 pain generally is neuropathic. (LLM, conf=0.86)

  Experimental — accuracy not yet measured

May combine two claims.

ACT has been found effective for adolescents and adults when delivered in weekly group sessions or in brief formats.

“Numerous researchers have found ACT to be effective among adolescents and adults when delivered via weekly group sessions [Butryn et al., 2011; Gauntlett-Gilbert et al., 2013] and when delivered in a brief format (e.g., in as little as four 1-hr sessions [Dahl et al., 2004]).”

• cites Butryn et al., 2011 sourceNot checked — no full text of the source
• cites Gauntlett-Gilbert et al., 2013 sourcePartly supported

  The Treatment section (p. 4) describes group ACT treatment for adolescents, and the Discussion (p. 8) reports positive outcomes. But the program was an intensive 3-week residential program with about 90 hours of treatment over 15 days, not weekly group sessions. The separate citations in the citing sentence address adults and brief formats. (LLM, conf=0.87)

  Experimental — accuracy not yet measured
• cites Dahl et al., 2004 sourceSupported

Probably not a substantial claim: may not pass S3 (load-bearing).

Initial research suggests that pain relief from mindfulness may involve decreased brain activation in pain-related areas through repeated practice.

“Initial research suggests that the mechanisms behind the pain relief may be related to decreased brain activation in areas associated with pain that occurs through repeated mindfulness practice [deCharms et al., 2005; Gard et al., 2012].”

• cites deCharms et al., 2005 sourcePartly supported

  The Results (page unavailable) link decreased activation in a pain-related region to lower pain ratings after training. But the training used real-time fMRI feedback, not repeated mindfulness practice; the paper does not establish a mechanism for mindfulness-related pain relief. (LLM, conf=0.88)

  Experimental — accuracy not yet measured
• cites Gard et al., 2012 sourceNot checked — no full text of the source

Probably not a substantial claim: may not pass S3 (load-bearing).

The finding that parent involvement may support adolescent adherence is consistent with prior research on parent involvement in adolescent interventions.

“This is consistent with prior research supporting the involvement of parents in interventions geared toward adolescents [Young et al., 2007; Stanger et al., 2013].”

• cites Young et al., 2007 sourceNot checked — no full text of the source
• cites Stanger et al., 2013 sourceSupported

May combine two claims.

60% of patients reported using mindfulness at least weekly and 60% reported using defusion techniques at least weekly.

“Sixty percent of patients reported using mindfulness at least weekly and 60% reported using defusion techniques at least weekly.”

Probably not a substantial claim: may not pass S3 (load-bearing).

Patient satisfaction ratings were not correlated with any outcome measures or adherence variables (all Ps > 0.05).

“Patient satisfaction ratings were not correlated with any outcome measures or adherence variables (all Ps > 0.05).”

May combine two claims.

Parent PAPIQ scores increased from baseline to 3 months, indicating greater acceptance of their child's pain, but the change was not statistically significant (P = 0.07).

“Furthermore, parent PAPIQ scores increased, indicating greater acceptance of their child’s pain, from baseline to 3 months, but this difference did not reach statistical significance (P = 0.07).”

May combine two claims.

71% of parents reported using mindfulness and 71% reported using defusion weekly or more; 71% reported encouraging their child to do a valued activity while in pain at least weekly.

“Most parents reported engaging in mindfulness (71%) and defusion (71%) weekly or more. Likewise, 71% reported encouraging their child to do a valued activity while in pain at least once a week.”

May combine two claims.

86% of enrolled patients attended all three workshop sessions; 83% of patients and 56% of parents who completed the workshop provided complete follow-up data.

“The majority of enrolled patients (86%) took part in all three workshop sessions, and 83% of patients and 56% of parents who completed the workshop provided complete follow-up data.”

May combine two claims.

Six of ten patients were taking less pain medication at follow-up than at baseline; three took fewer medications, two took a lower dose, and one took both fewer medications and a lower dose.

“Six of the ten patients were taking less pain medication at follow-up compared to baseline; of these, three were taking fewer medications, two were taking a lower dose, and one was taking both fewer medications and a lower dose.”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

Two patients were taking more medications and one increased her dose; one patient took no medications at either time point.

“Two patients were taking more medications and one increased the dose of her current medication. One patient was not taking any medications at either time point.”

Probably not a substantial claim: may not pass S3 (load-bearing).

Mean age of the ten completers was 16.9 years (SD = 2.9, range 12-20).

“The mean age of the ten patients who completed the study was 16.9 years (SD = 2.9, range 12–20 years).”

Probably not a substantial claim: may not pass S3 (load-bearing).

Nine mothers and three fathers completed the workshop.

“Regarding parents, nine mothers and three fathers completed the workshop.”

May combine two claims.

Only 64% of eligible families agreed to participate, mainly because of difficulty scheduling a trip to the research facility.

“Finally, only 64% of eligible families agreed to participate, mainly due to difficulty scheduling a trip to the research facility.”

Probably not a substantial claim: may not pass S3 (load-bearing).

Patient age, gender, and parent education were unrelated to primary study variables.

“Patient age, gender, and parent education were unrelated to primary study variables.”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

One mother could not be reached for the 1-month booster session, but her 3-month data were included.

“Also, we were unable to reach one mother for the 1-month booster session, but included her 3-month data in the analyses.”

Probably not a substantial claim: may not pass S3 (load-bearing).

Eight of ten patients were taking pain medication regularly at baseline.

“Eight of the ten patients were taking pain medication regularly at the baseline evaluation.”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

All patients were enrolled on an NF1 natural history study at the center, and none were on active NF1 treatment during the study.

“All of the patients were enrolled on an NF1 natural history study at our center; none of the youth were on active treatment for their NF1 (e.g., for tumor shrinkage) during the course of their participation on the current study.”

Probably not a substantial claim: may not pass S3 (load-bearing).

Mean parent education was 14.1 years (SD = 1.7, range 12-17).

“The mean level of parent education was 14.1 years (SD = 1.7, range 12–17 years).”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

Six females and six males completed the patient intervention; two did not return 3-month questionnaires, so data for the 10 patients who completed all time points are presented.

“Six females and six males completed the patient intervention. Of these, two did not return their 3-month questionnaires, so data for the 10 patients who completed all time points are presented in the sections that follow.”

May combine two claims.

Among patients with complete data, 20% had a learning disability, 20% had ADHD, and 30% had both.

“Among the patients in our study with complete data, most had been diagnosed either with a learning disability (20%), attention deficit hyperactivity disorder (ADHD; 20%), or both (30%).”

May combine two claims.

ACT is an empirically supported treatment for chronic pain that helps people focus on valued relationships and activities.

“Acceptance and Commitment Therapy (ACT), an empirically supported method for addressing chronic pain, helps individuals re-focus on valued relationships and activities.”

• Also stated in: § INTRODUCTION · plain

  “Acceptance and Commitment Therapy (ACT [Hayes et al., 1999]) is a psychological treatment that has been used to minimize pain interference and maximize quality of life (QOL) among adolescents and adults with chronic pain [Wicksell et al., 2009; Buhrman et al., 2013].”

• cites Hayes et al., 1999Not matched

  Couldn’t be matched to a paper in Crossref, so it can’t be checked.

• cites Wicksell et al., 2009 sourceNot checked — no full text of the source
• cites Buhrman et al., 2013 sourceNot checked — no full text of the source

May combine two claims.

The small sample size prevented subgroup analyses, so it is unknown whether the intervention worked differently by pain level, age, or maternal versus paternal participation.

“This prevented our ability to do subgroup analyses, so we do not know if the intervention worked differently for patients with varying levels of pain or of different ages (younger vs. older adolescents), or regarding maternal versus paternal participation.”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

Reduced pain intensity is not a goal of ACT but is not an uncommon finding in ACT studies.

“While this is not a goal of ACT, it is not an uncommon finding [Wicksell et al., 2009; McCracken and Gutierrez-Martinez, 2011; Thorsell et al., 2011].”

• cites Wicksell et al., 2009 sourceNot checked — no full text of the source
• cites McCracken and Gutierrez-Martinez, 2011 sourceNot checked — no full text of the source
• cites Thorsell et al., 2011 sourceNot checked — no full text of the source

Probably not a substantial claim: may not pass S3 (load-bearing).

Chronic pain is commonly reported in people with NF1 and plexiform neurofibromas.

“Chronic pain is commonly reported among individuals with NF1 and plexiform neurofibroma tumors (PNs).”

• Also stated in: § INTRODUCTION · plain

  “PNs are benign, sometimes painful tumors comprised of a proliferation of cells in the nerve sheath present in one-third to over half of people with NF1 [Tucker et al., 2009; Jett and Friedman, 2010].”

• cites Tucker et al., 2009 sourceNot checked — no full text of the source
• cites Jett and Friedman, 2010 sourceNot checked — no full text of the source

May combine two claims.

Parent participation in a parallel workshop may have contributed to patients' adherence to ACT skills at home, and vice versa.

“Having the parents participate in a parallel workshop so that they could support their child in practicing ACT-related skills may have contributed to the patient’s adherence to the intervention at home (and vice versa).”

May combine two claims.

The absence of change on functional disability may be because the PII measures pain interference while the FDI measures actual abilities, so limitations may relate less to pain than to other factors such as tumor location or size.

“We did not see similar changes on the measure of functional disability (FDI), possibly because the PII specifically asks respondents to consider how much pain has interfered with activities, whereas the FDI asks about actual abilities; thus, limitations may relate less to pain than other factors (e.g., tumor location or size).”

Probably not a substantial claim: may not pass S3 (load-bearing).

People engaged in values-based activities tend to report lower pain intensity.

“In addition, individuals who are engaged in values-based activities tend to report less pain intensity [McCracken et al., 2010b].”

• cites McCracken et al., 2010bNot matched

  Couldn’t be matched to a paper in Crossref, so it can’t be checked.

May combine two claims.

Adherence rates were fairly strong and comparable to other acceptance-based treatment protocols.

“The rates of adherence to the intervention were fairly strong, and in line with other acceptance-based treatment protocols [Woods et al., 2006; Goodwin et al., 2012].”

• cites Woods et al., 2006 sourceNot checked — no full text of the source
• cites Goodwin et al., 2012 sourceNot checked — no full text of the source

May combine two claims.

The 2-day small-group ACT workshops appear feasible and effective for adolescents and young adults with NF1 and chronic pain.

“Based on results of this pilot study, the 2-day, small group ACT workshops seem to be a feasible and effective intervention for AYA with NF1 and chronic pain.”

May combine two claims.

Patients and parents liked participating in the study, which supports feasibility.

“Overall, patients and parents liked participating in the study, which is encouraging with respect to feasibility.”

May combine two claims.

Because all 10 patients had PNs, the results may not represent the entire population of AYA with NF1.

“Since all 10 of our patients had PNs, results may not be representative of the entire population of AYA with NF1.”

May combine two claims.

Although the change in parents' acceptance of their child's pain was not statistically significant, the raw pre-post difference may be viewed with cautious optimism.

“Although changes in parents’ acceptance of their child’s pain did not reach statistical significance, the raw pre-post differences may be viewed with cautious optimism.”

May combine two claims.

Despite cognitive diagnoses, most participants appeared to understand the concepts and benefit from the intervention, as shown by lower mean pain interference and pain intensity scores at follow-up.

“Despite this fact, most participants seemed to understand the concepts introduced and were able to benefit from the intervention, as evidenced by lower group mean scores on the pain interference and pain intensity measures at the follow-up assessment.”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

Caregivers of pediatric medical patients have poorer psychosocial outcomes and quality of life than population norms, while parental acceptance of adolescent cancer pain is linked to better psychological wellbeing.

“Caregivers of pediatric medical patients have been found to have poorer psychosocial outcomes and QOL compared to population norms [Klassen et al., 2008], whereas parental acceptance of their adolescent’s cancer pain has been linked to better psychological wellbeing [Gauthier et al., 2009].”

• cites Klassen et al., 2008 sourceNot checked — no full text of the source
• cites Gauthier et al., 2009 sourceNot checked — no full text of the source

Classified as probably not a substantial claim.

Validated questionnaires assessed pain interference, pain intensity, functional disability, pain acceptance, pain-related anxiety, depression, health-related quality of life, parent psychological distress, and disease severity.

“These validated measures assessed domains of pain interference, pain intensity, functional disability, pain acceptance, pain-related anxiety, depression, health-related quality of life, parent psychological distress, and disease severity (completed by a nurse practitioner).”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

The cohort may be more motivated or higher functioning than the average family with a child with NF1 because families traveled from across the country to participate.

“Moreover, our cohort should be considered unique with respect to the fact that these families travel from all over the country in order to participate in a research study at this institution; as such, they may be more motivated (possibly due to more severe disease) or may be higher functioning than the average family with a child with NF1.”

Probably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.

The study procedures were approved by the research site's Institutional Review Board and registered with clinicaltrials.gov.

“The research site’s Institutional Review Board approved study procedures (registered with clinicaltrials.gov, #).”

Probably not a substantial claim: may not pass S3 (load-bearing).

The ACT workshop content was adapted from manualized ACT interventions.

“Workshop content was adapted from manualized ACT interventions outlined in books [Hayes et al., 1999; Dahl et al., 2005] and articles [Wicksell et al., 2007; Vowles et al., 2009; Masuda et al., 2011].”

• cites Hayes et al., 1999Not matched

  Couldn’t be matched to a paper in Crossref, so it can’t be checked.

• cites Dahl et al., 2005Not matched

  Couldn’t be matched to a paper in Crossref, so it can’t be checked.

• cites Wicksell et al., 2007 sourceNot checked — no full text of the source

May combine two claims.

ACT may help parents increase their acceptance of their child's pain, but larger randomized studies are needed to confirm this tentative finding.

“It also may help parents increase their acceptance of their child’s pain, although randomized studies with larger samples are needed to confirm these tentative findings.”