Appl Psychophysiol Biofeedback (2014) 39:287–291 DOI 10.1007/s10484-014-9261-x
HRV Biofeedback for Pediatric Irritable Bowel Syndrome and Functional Abdominal Pain: A Clinical Replication Series Mark J. Stern • Robert A. F. Guiles Richard Gevirtz
•
Published online: 2 October 2014 Ó Springer Science+Business Media New York 2014
Abstract Irritable bowel syndrome (IBS) and Functional Abdominal Pain (FAP) are among the most commonly reported Functional Gastrointestinal Disorders. Both have been associated with varying autonomic dysregulation. Heart Rate Variability Biofeedback (HRVB) has recently begun to show efficacy in the treatment of both IBS and FAP. The purpose of this multiple clinical replication series was to analyze the clinical outcomes of utilizing HRVB in a clinical setting. Archival data of twenty-seven consecutive pediatric outpatients diagnosed with IBS or FAP who received HRVB were analyzed. Clinical outcomes were self-report and categorized as full or remission with patient satisfaction, or no improvement. Qualitative reports of patient experiences were also noted. Full remission was achieved by 69.2 % and partial remission was achieved by 30.8 % of IBS patients. Full remission was achieved by 63.6 % and partial remission was achieved by 36.4 % of FAP patients. No patients in either group did not improve to a level of patient satisfaction or [50 %. Patient’s commonly reported feeling validated in their discomfort as a result of psychophysiological education. Results suggest that HRVB is a promising intervention for pediatric outpatients with IBS or FAP. Randomized controlled trials are necessary to accurately determine clinical efficacy of HRVB in the treatment of IBS and FAP. Keywords Clinical replication series Heart rate variability Biofeedback Irritable bowel syndrome Functional abdominal pain
M. J. Stern R. A. F. Guiles R. Gevirtz (&) Clinical Psychology PhD Program, California School of Professional Psychology, 10455 Pomerado Road, San Diego, CA 92131, USA e-mail: [email protected]; [email protected]
Introduction Functional gastrointestinal disorders (FGIDs) are chronic or recurring medical conditions that are largely unexplained by current structural or biochemical assessments (Drossman 2006; McOmber and Shulman 2008; Palsson and Whitehead 2013). FGIDs are associated with impaired quality of life and vast economic burdens, both individually and societally (Akehurst et al. 2002; Koloski et al. 2000; Maxion-Bergemann et al. 2006). Based on the ROME III criteria, the largest US national household survey on FGID prevalence to date (n = 5,430), researchers found that 69 % met criteria for at least one FGID (Drossman et al. 1993). Functional bowel disorders, such as Irritable Bowel Syndrome (IBS), are the most commonly reported FGIDs (41 and 28 %, respectively) (Thompson et al. 2002). IBS, alone, accounts for 25 % of all GI disorders seen by GI specialists and up to 12 % seen by primary care physicians (Schuster 2001). Median prevalence for pediatric Functional Abdominal Pain (FAP) is reported to be 8.4 % of school aged children (McOmber and Shulman 2008). Despite advances in medical and pharmaceutical interventions, treatment largely remains insufficient (Palsson and Whitehead 2013). The psychological comorbidity associated with FGIDs as well as the efficacy of psychological interventions are well documented (Drossman 2006; Palsson and Whitehead 2013), but the mechanisms of action are still reported to be unknown (Brandt et al. 2009). Defining FGIDs by the absence of identified disease biomarkers, ignores physiological dysfunction of ‘‘brain-gut’’ interaction associated with FGIDs. It is expected that interventions targeting the physiological aspects of these disorders would promote stronger clinical outcomes.
123
288
Irritable Bowel Syndrome
Appl Psychophysiol Biofeedback (2014) 39:287–291
whether sympathetic dysfunction is central or peripheral in origin (Chelimsky et al. 2001). There appears to be a neural reduction in sympathetic signals and increased resting parasympathetic activity (Jørgensen et al. 1993), possibly suggesting a lower central threshold for afferent sympathetic changes. Though Puzanovova et al. (2009) found no difference in vagal tone at rest between FAP and healthy controls, they did find heightened vagal reactivity to successful trials during cognitive tasks. Interestingly, Sowder et al. (2010) found that significant pain reduction in patients with FAP was mediated by gains in vagal tone through HRVB.
Irritable Bowel Syndrome (IBS) is the most common functional gastrointestinal disorder (Thompson et al. 2002), characterized by abdominal pain/discomfort associated with altered bowel patterns in the absence of organic biomarkers of disease (Brandt et al. 2009; Drossman 2006). IBS is commonly associated with autonomic dysregulation, as reflected by low Heart Rate Variability (HRV), sympathetic dominance and/or poor vagal tone at rest (Heitkemper et al. 1998; Karling et al. 1998; Mazur et al. 2007; Pellissier et al. 2010; Orr et al. 2000) as well as stress reactivity, orthostatic, or hand grip tests (Adeyemi et al. 1999; McAllister et al. 1990; Waring et al. 2004). This is consistent with research that shows healthy gastric and colonic motility rely on autonomic regulation, particularly dominated by parasympathetic activity (Goyal and Hirano 1996; Ja¨nig 2006). There is also evidence that distinguishing sympathetic or parasympathetic dysfunction may differentiate IBS-Constipation type from IBS-Diarrhea type (Aggarwal et al. 1994). The autonomic dysregulatory component of IBS suggests that restoring ANS regulation should improve GI symptoms. Though research investigating HRV Biofeedback (HRVB) with IBS is relatively novel, evidence of potential efficacy is beginning to appear. HRVB was shown to improve symptoms of IBS equivalently to hypnotherapy (Dobbin et al. 2013), a previously established efficacious treatment for IBS (Lee et al. 2014). However, the sample was exclusively female adults with strict exclusion criteria (for example, no medication that may impact cardiac autonomic tone). Though this provides strong internal validity, it is difficult to generalize the clinical utility among non-discriminated, consecutive referrals.
HRV Biofeedback has been shown to directly improve autonomic regulation and restore vagal tone (Gevirtz 2000; Lehrer et al. 2000). The autonomic dysregulatory component of IBS and FAP suggests that restoring ANS regulation should improve GI symptoms. Though research investigating HRV Biofeedback (HRVB) with FGIDs is relatively novel, evidence, as mentioned above, is beginning to appear. The purpose of this study was to evaluate the clinical utility of Heart Rate Variability Biofeedback (HRVB) in the treatment of IBS and FAP among pediatric outpatients. This clinical replication series is the first to report clinical outcomes for HRVB with pediatric IBS. Therefore a clinical replication series design of archival data was implemented to explore clinical outcomes in a clinical patient population. The advantage of the clinical replication series is to determine generality by normally delivering an intervention in an actual field setting (Barlow et al. 1984).
Functional Abdominal Pain
Methods
According to the Rome III diagnostic criteria, FAP is marked by episodic or recurrent abdominal pain without meeting criteria for other FGIDs and FAP Syndrome (FAPS) is marked by continuous or nearly continuous abdominal pain with impairment in daily functioning (Drossman 2006). FAP and FAPS pathophysiology is unique from other FGIDs in that GI functioning is relatively undisturbed (Sperber and Drossman 2010). It is considered largely a disorder of central and peripheral nervous system integration (Grover and Drossman 2010). There is an abnormal perception of pain to normal gut functioning (Nozu and Okumura 2011). In fact, patients with FAP have been shown not do differ in pain perception during rectal pain threshold testing. Individual differences on varying autonomic tests make it difficult to determine
Participants
123
Objective
Twenty-Seven consecutive pediatric patients, (age range 6–17 years) diagnosed with IBS or FAP by their gastroenterologist were referred for HRVB. All patients presented to their physician for GI complaints. Each patient was randomly assigned to be treated by one of three supervised clinical psychology doctoral practicum students. Written informed consent was obtained prior to treatment. One IBS patient was excluded from this analysis due to comorbid inflammatory bowel disease. Two IBS patients dropped out after the initial intake session. There were dropouts or exclusions from the FAP referrals. A total of 13 IBS and 11 FAP patients, were treated with HRVB for IBS symptoms (see Table 1 for patient characteristics).
Appl Psychophysiol Biofeedback (2014) 39:287–291
289
Table 1 Patient characteristics for both IBS and FAP
Table 2 Remission rates by diagnosis
Characteristic
IBS
FAP
Sample
n = 13
n = 11
Diagnosis IBS
Subtype
Full remission (%) 69.20
Partial remission (%)
Not improved (%)
30.80
0.00
IBS-D
n=3
N/A
Subtype
IBS-C
n=4
N/A
IBS-D
100.00
0.00
0.00
N/A
IBS-C
50.00
50.00
0.00
7:6
IBS-M
60.00
40.00
0.00
63.60
36.40
0.00
IBS-M Gender F:M
n=6 7:4
Age
10.69 (3.401)
12.18 (3.06)
FAP
No. sessions
8.15 (3.997)
10.36 (4.388)
Remission rates were determined by patients’ self-reports
No. session range
3–18
4–19
Heart Rate Variability Biofeedback In HRVB, real-time heart rate and respiration are noninvasively measured and displayed on a monitor for patients to train their autonomic nervous system through diaphragmatic breathing at their resonant frequency breath rate. Other physiological measures displayed during sessions included skin conductance and skin temperature as supplementary references for real-time autonomic changes. All physiological monitoring and biofeedback training was done with J&J Engineering C2? hardware and J&J Engineering USE3 software. All patients began with appropriate paperwork, including the informed consent, and extensive intake regarding their symptom and psychosocial history. Pediatric friendly psychophysiological education about the ‘‘brain-gut’’ interaction was also provided during the intake session. Psychophysiological assessment was visually analyzed and utilized in the education component, reviewing images of their autonomic activity at rest, during a mild cognitive stressor, and during recovery. During rest, patients were asked to sit quietly for 5 min. As the mild stressor, patients were asked to count backwards by 7 as quickly as they can, while being moderately pressed for speed by the clinician. Recovery was again monitored for 5 min. Each of these three components was clinically utilized as psychophysiological education tools. Data for these components were not recorded. HRVB began at the second session and consisted of several phases. Each session consisted of 30 min of biofeedback for an average of 8 sessions. Phase I: Diaphragmatic breathing was taught to the patient using respiratory biofeedback and clinical monitoring. Phase II: Resonant frequency breath rate was established for each patient. Phase III: Patients were instructed to breathe at their resonant frequency, utilizing the feedback from the spectral and time domain displays for heart rate variability as well as respiration. Training focused on increasing peak valley
differences in heart rate, beat to beat, to maximize the low frequency amplitude as a marker of sympathovagal balance and maintaining relative respiratory pace. Patients were also instructed to breathe at home for minimum of 20 min per day using either computer/phone applications, such as StressEraser, or music scales (BreathSync) designed to simulate their resonant frequency breathe rate. Outcome measures were symptom frequency and severity by self-report. A priori, clinical criteria for therapy termination was developed based on symptom reporting. Full remission was characterized as having no symptoms for at least two full weeks. Partial remission was characterized as having a 50 % improvement in symptoms (consistent with Neff and Blanchard 1987) and patient reporting no impairment in daily functioning for at least two full weeks.
Results Full remission was achieved by 69.2 % and partial remission was achieved by 30.8 % of the IBS patients. IBS patients received an average of 8.15 (SD = 3.997), with a range of 3–18 HRVB sessions. Exploratory analysis of IBS subtypes showed that there was no statistical difference in remission rates between IBS-Diarrhea, IBS-Constipation, and IBS-Mixed types (See Table 2 for remission rates by subtype). Full remission was achieved by 63.6 % and partial remission was achieved by 36.4 % of FAP patients. FAP patients received an average of 10.36 (SD = 4.388), with a range of 4–19 HRVB sessions. There were no patients in either group that did not improve to a level of patient satisfaction or [50 % by self-report. See Table 2 for remission rates. There was no group difference in remission rates between IBS or FAP. Neither age nor gender predicted difference between full or partial remission in either IBS or FAP. Analysis of gender by number of sessions revealed a trend (r = -.563, p = .076), thought non-significant, indicating that there may be a relationship whereby females require a greater number of sessions.
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Moderator analyses must be viewed with caution due to the small sample size. Qualitatively, over two-thirds of IBS and FAP patients’ experiences appeared to share overlapping themes throughout the intervention. At the initial intake, patients commonly expressed concern and confusion over their referral to a mental health practitioner. After psychophysiological education, most patients reported feeling validated in the ‘‘reality’’ of their symptoms and felt confident in the model fit of treatment to diagnosis. Treatment compliance1 with homework commonly waned as patients began to feel improvement, but improved after a session that included maintenance education. Post treatment, patients in both diagnostic groups expressed overall satisfaction with both their clinical outcomes and the intervention as a whole.
Discussion Results from this multiple clinical replication series support the clinical utility of HRVB in the treatment of IBS and FAP in pediatric outpatients. Significant portions of both IBS and FAP patients improved to a level of full remission and clinically significant partial remission ([50 %) by self-report. IBS had a higher percentage of patients that reached full remission than FAP, though not significant. A large portion of FAP patients also achieved clinically significant improvements. There was no difference in HRVB outcomes between across IBS sub-types. Neither gender nor age predicted the difference between full or partial remission at post treatment. Male patients may respond quicker to HRVB in both IBS and FAP, requiring less sessions overall. However, all moderator analyses should be considered exploratory due to the small sample size in each cell. As such, studies with greater sample size are needed to confirm if the gender by session number interaction trend would reach significance. The qualitative reports support that psychophysiological interventions may help validate patients’ experiences, creating additional interest, patient satisfaction, and compliance. HRVB shows promise for both symptom reduction and patient satisfaction. Functional GI Disorders are in need of more fitting and cost effective treatments. There is growing evidence that symptoms of IBS and FAP are psychophyisiological dysfunctions relating to autonomic and central dysregulation (Chelimsky et al. 2001; Grover and Drossman 2010; Heitkemper et al. 1998; Karling et al. 1998; Mazur et al. 2007; Nozu and Okumura 2011; Pellissier et al. 2010; Orr et al. 2000). The mechanism of action behind HRVB is hypothesized to be the restoration of autonomic regulation (Gevirtz 2000; Lehrer et al. 2000). If controlled trials
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replicate the positive results reported here, this theory would be supported. The low number of treatment sessions required to achieve clinical significance, especially in IBS, may also show HRVB to be the most cost-effective treatment available. This study is limited by small sample size within each FGID and single self-report measure for clinical outcomes. HRV data during baseline and stress task were not recorded, limiting mediational interpretation. Treatment compliance was also not systematically assessed. These factors should be incorporated in future studies. There is also limited internal validity in the nature of clinical replication series designs since there is no control arm to ensure that improvements are significantly different from that of usual care or other treatment modalities. Clinical outcomes were only assessed 2 weeks post treatment. Follow-up assessments at a minimum of 3 months should be included in future studies. Additional randomized controlled trials with both active and passive controls as well as standardized measures are recommended to further evaluate the potential effectiveness of HRVB in the treatment of IBS and FAP. Future research should also investigate potential moderators for treatment success, number of sessions, and possible differential autonomic dysfunctions between IBS and FAP.
References Adeyemi, E., Desai, K., Towsey, M., & Ghista, D. (1999). Characterization of autonomic dysfunction in patients with irritable bowel syndrome by means of heart rate variability studies. The American Journal of Gastroenterology, 94(3), 816–823. Aggarwal, A., Cutts, T., Abell, T., Cardoso, S., Familoni, B., Bremer, J., et al. (1994). Predominant symptoms in irritable bowel syndrome correlate with specific autonomic nervous system abnormalities. Gastroenterology, 106(4), 945–950. Akehurst, R. L., Brazier, J. E., Mathers, N. N., O’Keefe, C. C., Kaltenthaler, E. E., & Morgan, A. A. (2002). Health-related quality of life and cost impact of irritable bowel syndrome in a UK primary care setting. Pharmacoeconomics, 20(7), 455–462. Barlow, D. H., Hayes, S. C., & Nelson, R. O. (1984). The scientist practitioner: Research and accountability in clinical and education settings. Elmsford: Pergamon Press. Brandt, L., Chey, W., Foxx-Orenstein, A., Schiller, L., Schoenfeld, P., & Spiegel, B. (2009). An evidence-based position statement on the management of irritable bowel syndrome. The American Journal of Gastroenterology, 104(Suppl 1), S1–S35. doi:10. 1038/ajg.2008.122. Chelimsky, G., Boyle, J., Tusing, L., & Chelimsky, T. (2001). Autonomic abnormalities in children with functional abdominal pain: Coincidence or etiology? Journal of Pediatric Gastroenterology and Nutrition, 33(1), 47–53. Dobbin, A., Dobbin, J., Ross, S., Graham, C., & Ford, M. (2013). Randomised controlled trial of brief intervention with biofeedback and hypnotherapy in patients with refractory irritable bowel syndrome. The Journal of the Royal College of Physicians of Edinburgh, 43(1), 15–23. doi:10.4997/JRCPE.2013.104.
Appl Psychophysiol Biofeedback (2014) 39:287–291 Drossman, D. (2006). The functional gastrointestinal disorders and the Rome III process. Gastroenterology, 130(5), 1377–1390. Drossman, D. A., Li, Z., Andruzzi, E., Temple, R. D., Talley, N. J., & Thompson, W. (1993). U.S. householder survey of functional gastrointestinal disorders. Digestive Diseases and Sciences, 38(9), 1569–1580. Gevirtz, R. (2000). Resonant frequency training to restore autonomic homeostasis for treatment of psycho physiologic disorders. Biofeedback, 27(1), 7–9. Goyal, R., & Hirano, I. (1996). The enteric nervous system. The New England Journal of Medicine, 334(17), 1106–1115. Grover, M., & Drossman, D. (2010). Functional abdominal pain. Current gastroenterology reports, 12(5), 391–398. doi:10.1007/ s11894-010-0125-0. Heitkemper, M., Burr, R., Jarrett, M., Hertig, V., Lustyk, M., & Bond, E. (1998). Evidence for autonomic nervous system imbalance in women with irritable bowel syndrome. Digestive Diseases and Sciences, 43(9), 2093–2098. Ja¨nig, W. (2006). The integrative action of the autonomic nervous system: Neurobiology of homeostasis. Cambridge: Cambridge University Press. Jørgensen, L., Christiansen, P., Raundahl, U., Ostgaard, S., Christensen, N., Fenger, M., et al. (1993). Autonomic nervous system function in patients with functional abdominal pain. An experimental study. Scandinavian Journal of Gastroenterology, 28(1), 63–68. Karling, P., Nyhlin, H., Wiklund, U., Sjo¨berg, M., Olofsson, B., & Bjerle, P. (1998). Spectral analysis of heart rate variability in patients with irritable bowel syndrome. Scandinavian Journal of Gastroenterology, 33(6), 572–576. Koloski, N., Talley, N., & Boyce, P. (2000). The impact of functional gastrointestinal disorders on quality of life. The American Journal of Gastroenterology, 95(1), 67–71. Lee, H., Choi, Y., & Choi, M. (2014). The efficacy of hypnotherapy in the treatment of irritable bowel syndrome: A systematic review and meta-analysis. Journal of Neurogastroenterology and Motility, 20(2), 152–162. doi:10.5056/jnm.2014.20.2.152. Lehrer, P. M., Vaschillo, E., & Vaschillo, B. (2000). Resonant frequency biofeedback training to increase cardiac variability: Rationale and manual for training. Applied Psychophysiology & Biofeedback, 25(3), 177–191. Maxion-Bergemann, S., Thielecke, F., Abel, F., & Bergemann, R. (2006). Costs of irritable bowel syndrome in the UK and US. Pharmacoeconomics, 24(1), 21–37. Mazur, M., Furgała, A., Jabłon´ski, K., Madroszkiewicz, D., Ciec´koMichalska, I., Bugajski, A., et al. (2007). Dysfunction of the autonomic nervous system activity is responsible for gastric myoelectric disturbances in the irritable bowel syndrome patients. Journal of Physiology and Pharmacology: An Official Journal of the Polish Physiological Society, 58(Suppl), 3131–3139. McAllister, C., McGrath, F., & Fielding, J. (1990). Altered skin temperature and electromyographic activity in the irritable bowel syndrome. Biomedicine & Pharmacotherapy, 44(8), 399–401.
291 McOmber, M. A., & Shulman, R. J. (2008). Pediatric functional gastrointestinal disorders. Nutrition in Clinical Practice, 23(3), 268–274. doi:10.1177/0884533608318671. Neff, D. F., & Blanchard, E. B. (1987). A multi-component treatment for irritable bowel syndrome. Behavior Therapy, 18(1), 70–83. doi:10.1016/S0005-7894(87)80052-7. Nozu, T., & Okumura, T. (2011). Visceral sensation and irritable bowel syndrome; with special reference to comparison with functional abdominal pain syndrome. Journal of Gastroenterology and Hepatology, 26, 122–127. doi:10.1111/j.1440-1746. 2011.06636.x. Orr, W., Elsenbruch, S., & Harnish, M. (2000). Autonomic regulation of cardiac function during sleep in patients with irritable bowel syndrome. The American Journal of Gastroenterology, 95(10), 2865–2871. Palsson, O., & Whitehead, W. (2013). Psychological treatments in functional gastrointestinal disorders: A primer for the gastroenterologist. Clinical Gastroenterology and Hepatology: The Official Clinical Practice Journal of the American Gastroenterological Association, 11(3), 208. doi:10.1016/j.cgh.2012.10.031. Pellissier, S., Dantzer, C., Canini, F., Mathieu, N., & Bonaz, B. (2010). Psychological adjustment and autonomic disturbances in inflammatory bowel diseases and irritable bowel syndrome. Psychoneuroendocrinology, 35(5), 653–662. doi:10.1016/j.psy neuen.2009.10.004. Puzanovova, M., Arbogast, P. G., Smith, C. A., Anderson, J., Diedrich, A., & Walker, L. S. (2009). Autonomic activity and somatic symptoms in response to success versus failure on a cognitive task: A comparison of chronic abdominal pain patients and well children. Journal of Psychosomatic Research, 67(3), 235–243. doi:10.1016/j.jpsychores.2009.02.007. Schuster, M. (2001). Defining and diagnosing irritable bowel syndrome. The American Journal of Managed Care, 7(8 Suppl), S246–S251. Sowder, E., Gevirtz, R., Shapiro, W., & Ebert, C. (2010). Restoration of vagal tone: A possible mechanism for functional abdominal pain. Applied Psychophysiol Biofeedback. Sperber, A., & Drossman, D. (2010). Functional abdominal pain syndrome: Constant or frequently recurring abdominal pain. The American Journal of Gastroenterology, 105(4), 770–774. doi:10. 1038/ajg.2010.68. Thompson, W., Irvine, E., Pare, P., Ferrazzi, S., & Rance, L. (2002). Functional gastrointestinal disorders in Canada: first populationbased survey using Rome II criteria with suggestions for improving the questionnaire. Digestive Diseases and Sciences, 47(1), 225–235. Waring, W., Chui, M., Japp, A., Nicol, E., & Ford, M. (2004). Autonomic cardiovascular responses are impaired in women with irritable bowel syndrome. Journal of Clinical Gastroenterology, 38(8), 658–663.
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HRV Biofeedback for Pediatric Irritable Bowel Syndrome and Functional Abdominal Pain: A Clinical Replication Series Mark J. Stern • Robert A. F. Guiles Richard Gevirtz
•
Published online: 2 October 2014 Ó Springer Science+Business Media New York 2014
Abstract Irritable bowel syndrome (IBS) and Functional Abdominal Pain (FAP) are among the most commonly reported Functional Gastrointestinal Disorders. Both have been associated with varying autonomic dysregulation. Heart Rate Variability Biofeedback (HRVB) has recently begun to show efficacy in the treatment of both IBS and FAP. The purpose of this multiple clinical replication series was to analyze the clinical outcomes of utilizing HRVB in a clinical setting. Archival data of twenty-seven consecutive pediatric outpatients diagnosed with IBS or FAP who received HRVB were analyzed. Clinical outcomes were self-report and categorized as full or remission with patient satisfaction, or no improvement. Qualitative reports of patient experiences were also noted. Full remission was achieved by 69.2 % and partial remission was achieved by 30.8 % of IBS patients. Full remission was achieved by 63.6 % and partial remission was achieved by 36.4 % of FAP patients. No patients in either group did not improve to a level of patient satisfaction or [50 %. Patient’s commonly reported feeling validated in their discomfort as a result of psychophysiological education. Results suggest that HRVB is a promising intervention for pediatric outpatients with IBS or FAP. Randomized controlled trials are necessary to accurately determine clinical efficacy of HRVB in the treatment of IBS and FAP. Keywords Clinical replication series Heart rate variability Biofeedback Irritable bowel syndrome Functional abdominal pain
M. J. Stern R. A. F. Guiles R. Gevirtz (&) Clinical Psychology PhD Program, California School of Professional Psychology, 10455 Pomerado Road, San Diego, CA 92131, USA e-mail: [email protected]; [email protected]
Introduction Functional gastrointestinal disorders (FGIDs) are chronic or recurring medical conditions that are largely unexplained by current structural or biochemical assessments (Drossman 2006; McOmber and Shulman 2008; Palsson and Whitehead 2013). FGIDs are associated with impaired quality of life and vast economic burdens, both individually and societally (Akehurst et al. 2002; Koloski et al. 2000; Maxion-Bergemann et al. 2006). Based on the ROME III criteria, the largest US national household survey on FGID prevalence to date (n = 5,430), researchers found that 69 % met criteria for at least one FGID (Drossman et al. 1993). Functional bowel disorders, such as Irritable Bowel Syndrome (IBS), are the most commonly reported FGIDs (41 and 28 %, respectively) (Thompson et al. 2002). IBS, alone, accounts for 25 % of all GI disorders seen by GI specialists and up to 12 % seen by primary care physicians (Schuster 2001). Median prevalence for pediatric Functional Abdominal Pain (FAP) is reported to be 8.4 % of school aged children (McOmber and Shulman 2008). Despite advances in medical and pharmaceutical interventions, treatment largely remains insufficient (Palsson and Whitehead 2013). The psychological comorbidity associated with FGIDs as well as the efficacy of psychological interventions are well documented (Drossman 2006; Palsson and Whitehead 2013), but the mechanisms of action are still reported to be unknown (Brandt et al. 2009). Defining FGIDs by the absence of identified disease biomarkers, ignores physiological dysfunction of ‘‘brain-gut’’ interaction associated with FGIDs. It is expected that interventions targeting the physiological aspects of these disorders would promote stronger clinical outcomes.
123
288
Irritable Bowel Syndrome
Appl Psychophysiol Biofeedback (2014) 39:287–291
whether sympathetic dysfunction is central or peripheral in origin (Chelimsky et al. 2001). There appears to be a neural reduction in sympathetic signals and increased resting parasympathetic activity (Jørgensen et al. 1993), possibly suggesting a lower central threshold for afferent sympathetic changes. Though Puzanovova et al. (2009) found no difference in vagal tone at rest between FAP and healthy controls, they did find heightened vagal reactivity to successful trials during cognitive tasks. Interestingly, Sowder et al. (2010) found that significant pain reduction in patients with FAP was mediated by gains in vagal tone through HRVB.
Irritable Bowel Syndrome (IBS) is the most common functional gastrointestinal disorder (Thompson et al. 2002), characterized by abdominal pain/discomfort associated with altered bowel patterns in the absence of organic biomarkers of disease (Brandt et al. 2009; Drossman 2006). IBS is commonly associated with autonomic dysregulation, as reflected by low Heart Rate Variability (HRV), sympathetic dominance and/or poor vagal tone at rest (Heitkemper et al. 1998; Karling et al. 1998; Mazur et al. 2007; Pellissier et al. 2010; Orr et al. 2000) as well as stress reactivity, orthostatic, or hand grip tests (Adeyemi et al. 1999; McAllister et al. 1990; Waring et al. 2004). This is consistent with research that shows healthy gastric and colonic motility rely on autonomic regulation, particularly dominated by parasympathetic activity (Goyal and Hirano 1996; Ja¨nig 2006). There is also evidence that distinguishing sympathetic or parasympathetic dysfunction may differentiate IBS-Constipation type from IBS-Diarrhea type (Aggarwal et al. 1994). The autonomic dysregulatory component of IBS suggests that restoring ANS regulation should improve GI symptoms. Though research investigating HRV Biofeedback (HRVB) with IBS is relatively novel, evidence of potential efficacy is beginning to appear. HRVB was shown to improve symptoms of IBS equivalently to hypnotherapy (Dobbin et al. 2013), a previously established efficacious treatment for IBS (Lee et al. 2014). However, the sample was exclusively female adults with strict exclusion criteria (for example, no medication that may impact cardiac autonomic tone). Though this provides strong internal validity, it is difficult to generalize the clinical utility among non-discriminated, consecutive referrals.
HRV Biofeedback has been shown to directly improve autonomic regulation and restore vagal tone (Gevirtz 2000; Lehrer et al. 2000). The autonomic dysregulatory component of IBS and FAP suggests that restoring ANS regulation should improve GI symptoms. Though research investigating HRV Biofeedback (HRVB) with FGIDs is relatively novel, evidence, as mentioned above, is beginning to appear. The purpose of this study was to evaluate the clinical utility of Heart Rate Variability Biofeedback (HRVB) in the treatment of IBS and FAP among pediatric outpatients. This clinical replication series is the first to report clinical outcomes for HRVB with pediatric IBS. Therefore a clinical replication series design of archival data was implemented to explore clinical outcomes in a clinical patient population. The advantage of the clinical replication series is to determine generality by normally delivering an intervention in an actual field setting (Barlow et al. 1984).
Functional Abdominal Pain
Methods
According to the Rome III diagnostic criteria, FAP is marked by episodic or recurrent abdominal pain without meeting criteria for other FGIDs and FAP Syndrome (FAPS) is marked by continuous or nearly continuous abdominal pain with impairment in daily functioning (Drossman 2006). FAP and FAPS pathophysiology is unique from other FGIDs in that GI functioning is relatively undisturbed (Sperber and Drossman 2010). It is considered largely a disorder of central and peripheral nervous system integration (Grover and Drossman 2010). There is an abnormal perception of pain to normal gut functioning (Nozu and Okumura 2011). In fact, patients with FAP have been shown not do differ in pain perception during rectal pain threshold testing. Individual differences on varying autonomic tests make it difficult to determine
Participants
123
Objective
Twenty-Seven consecutive pediatric patients, (age range 6–17 years) diagnosed with IBS or FAP by their gastroenterologist were referred for HRVB. All patients presented to their physician for GI complaints. Each patient was randomly assigned to be treated by one of three supervised clinical psychology doctoral practicum students. Written informed consent was obtained prior to treatment. One IBS patient was excluded from this analysis due to comorbid inflammatory bowel disease. Two IBS patients dropped out after the initial intake session. There were dropouts or exclusions from the FAP referrals. A total of 13 IBS and 11 FAP patients, were treated with HRVB for IBS symptoms (see Table 1 for patient characteristics).
Appl Psychophysiol Biofeedback (2014) 39:287–291
289
Table 1 Patient characteristics for both IBS and FAP
Table 2 Remission rates by diagnosis
Characteristic
IBS
FAP
Sample
n = 13
n = 11
Diagnosis IBS
Subtype
Full remission (%) 69.20
Partial remission (%)
Not improved (%)
30.80
0.00
IBS-D
n=3
N/A
Subtype
IBS-C
n=4
N/A
IBS-D
100.00
0.00
0.00
N/A
IBS-C
50.00
50.00
0.00
7:6
IBS-M
60.00
40.00
0.00
63.60
36.40
0.00
IBS-M Gender F:M
n=6 7:4
Age
10.69 (3.401)
12.18 (3.06)
FAP
No. sessions
8.15 (3.997)
10.36 (4.388)
Remission rates were determined by patients’ self-reports
No. session range
3–18
4–19
Heart Rate Variability Biofeedback In HRVB, real-time heart rate and respiration are noninvasively measured and displayed on a monitor for patients to train their autonomic nervous system through diaphragmatic breathing at their resonant frequency breath rate. Other physiological measures displayed during sessions included skin conductance and skin temperature as supplementary references for real-time autonomic changes. All physiological monitoring and biofeedback training was done with J&J Engineering C2? hardware and J&J Engineering USE3 software. All patients began with appropriate paperwork, including the informed consent, and extensive intake regarding their symptom and psychosocial history. Pediatric friendly psychophysiological education about the ‘‘brain-gut’’ interaction was also provided during the intake session. Psychophysiological assessment was visually analyzed and utilized in the education component, reviewing images of their autonomic activity at rest, during a mild cognitive stressor, and during recovery. During rest, patients were asked to sit quietly for 5 min. As the mild stressor, patients were asked to count backwards by 7 as quickly as they can, while being moderately pressed for speed by the clinician. Recovery was again monitored for 5 min. Each of these three components was clinically utilized as psychophysiological education tools. Data for these components were not recorded. HRVB began at the second session and consisted of several phases. Each session consisted of 30 min of biofeedback for an average of 8 sessions. Phase I: Diaphragmatic breathing was taught to the patient using respiratory biofeedback and clinical monitoring. Phase II: Resonant frequency breath rate was established for each patient. Phase III: Patients were instructed to breathe at their resonant frequency, utilizing the feedback from the spectral and time domain displays for heart rate variability as well as respiration. Training focused on increasing peak valley
differences in heart rate, beat to beat, to maximize the low frequency amplitude as a marker of sympathovagal balance and maintaining relative respiratory pace. Patients were also instructed to breathe at home for minimum of 20 min per day using either computer/phone applications, such as StressEraser, or music scales (BreathSync) designed to simulate their resonant frequency breathe rate. Outcome measures were symptom frequency and severity by self-report. A priori, clinical criteria for therapy termination was developed based on symptom reporting. Full remission was characterized as having no symptoms for at least two full weeks. Partial remission was characterized as having a 50 % improvement in symptoms (consistent with Neff and Blanchard 1987) and patient reporting no impairment in daily functioning for at least two full weeks.
Results Full remission was achieved by 69.2 % and partial remission was achieved by 30.8 % of the IBS patients. IBS patients received an average of 8.15 (SD = 3.997), with a range of 3–18 HRVB sessions. Exploratory analysis of IBS subtypes showed that there was no statistical difference in remission rates between IBS-Diarrhea, IBS-Constipation, and IBS-Mixed types (See Table 2 for remission rates by subtype). Full remission was achieved by 63.6 % and partial remission was achieved by 36.4 % of FAP patients. FAP patients received an average of 10.36 (SD = 4.388), with a range of 4–19 HRVB sessions. There were no patients in either group that did not improve to a level of patient satisfaction or [50 % by self-report. See Table 2 for remission rates. There was no group difference in remission rates between IBS or FAP. Neither age nor gender predicted difference between full or partial remission in either IBS or FAP. Analysis of gender by number of sessions revealed a trend (r = -.563, p = .076), thought non-significant, indicating that there may be a relationship whereby females require a greater number of sessions.
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Moderator analyses must be viewed with caution due to the small sample size. Qualitatively, over two-thirds of IBS and FAP patients’ experiences appeared to share overlapping themes throughout the intervention. At the initial intake, patients commonly expressed concern and confusion over their referral to a mental health practitioner. After psychophysiological education, most patients reported feeling validated in the ‘‘reality’’ of their symptoms and felt confident in the model fit of treatment to diagnosis. Treatment compliance1 with homework commonly waned as patients began to feel improvement, but improved after a session that included maintenance education. Post treatment, patients in both diagnostic groups expressed overall satisfaction with both their clinical outcomes and the intervention as a whole.
Discussion Results from this multiple clinical replication series support the clinical utility of HRVB in the treatment of IBS and FAP in pediatric outpatients. Significant portions of both IBS and FAP patients improved to a level of full remission and clinically significant partial remission ([50 %) by self-report. IBS had a higher percentage of patients that reached full remission than FAP, though not significant. A large portion of FAP patients also achieved clinically significant improvements. There was no difference in HRVB outcomes between across IBS sub-types. Neither gender nor age predicted the difference between full or partial remission at post treatment. Male patients may respond quicker to HRVB in both IBS and FAP, requiring less sessions overall. However, all moderator analyses should be considered exploratory due to the small sample size in each cell. As such, studies with greater sample size are needed to confirm if the gender by session number interaction trend would reach significance. The qualitative reports support that psychophysiological interventions may help validate patients’ experiences, creating additional interest, patient satisfaction, and compliance. HRVB shows promise for both symptom reduction and patient satisfaction. Functional GI Disorders are in need of more fitting and cost effective treatments. There is growing evidence that symptoms of IBS and FAP are psychophyisiological dysfunctions relating to autonomic and central dysregulation (Chelimsky et al. 2001; Grover and Drossman 2010; Heitkemper et al. 1998; Karling et al. 1998; Mazur et al. 2007; Nozu and Okumura 2011; Pellissier et al. 2010; Orr et al. 2000). The mechanism of action behind HRVB is hypothesized to be the restoration of autonomic regulation (Gevirtz 2000; Lehrer et al. 2000). If controlled trials
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replicate the positive results reported here, this theory would be supported. The low number of treatment sessions required to achieve clinical significance, especially in IBS, may also show HRVB to be the most cost-effective treatment available. This study is limited by small sample size within each FGID and single self-report measure for clinical outcomes. HRV data during baseline and stress task were not recorded, limiting mediational interpretation. Treatment compliance was also not systematically assessed. These factors should be incorporated in future studies. There is also limited internal validity in the nature of clinical replication series designs since there is no control arm to ensure that improvements are significantly different from that of usual care or other treatment modalities. Clinical outcomes were only assessed 2 weeks post treatment. Follow-up assessments at a minimum of 3 months should be included in future studies. Additional randomized controlled trials with both active and passive controls as well as standardized measures are recommended to further evaluate the potential effectiveness of HRVB in the treatment of IBS and FAP. Future research should also investigate potential moderators for treatment success, number of sessions, and possible differential autonomic dysfunctions between IBS and FAP.
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