Jiscs.com

Title: Counterstrain manipulation in the treatment of Restless
Legs Syndrome: a pilot single-blind randomised controlled trial;
the CARL Trial.
Peters, T 1, MacDonald, R* 1, Leach, CMJ 2
1 London College of Osteopathic Medicine, Marylebone, London, UK
2 Clinical Research Centre for Health Professions, University of Brighton, UK.
CORRESPONDENCE : R S MacDonald, LCOM, 8, Boston Place, NW1 6QH, UK. [email protected], Tel: 0207 262 1128
KEYWORDS: musculoskeletal manipulations, osteopathic medicine, Counterstrain, restless legs
syndrome, Randomised controlled trial
ABSTRACT
Objective
A single-blind randomised controlled trial was conducted to test the efficacy of Longden’s
counterstrain technique for restless legs syndrome (RLS) and the feasibility of the trial methodology.

Methods
Participants were adults with moderate to severe and persistent RLS, randomized to receive either
active or control intervention. The control intervention (B) involved Counterstrain manipulation applied
to the lower half of the body. The active intervention (A) was identical to the control intervention plus
specific modifications to treat RLS as described by Longden. The success of blinding of participants
was confirmed by a questionnaire.
Results
Thirty-nine patients entered the trial, 20 assigned to Group A and 19 to Group B. All patients were
included in the intention to treat analysis. The primary outcome measure, the change on the
International Restless Legs Scale (IRLS) total score at six weeks, showed a statistically significant
difference of 8·06 points (95% CI 3·15 - 12·96) between groups. This represented an improvement of
42·2% in the active group compared to 8·7% in the controls. No adverse effects were reported.
Conclusions
Longden’s RLS-specific Counterstrain treatment had a clinically important effect at six weeks. Trials
of longer term effects and comparison with the standard drug regimes are now required.

Introduction
Restless Legs Syndrome (RLS), described by Ekbom in 1945, is a condition characterized by a
strong inclination to move limbs, usually the legs, that comes on most frequently in bed at night but
may occur at rest while sitting. Movement temporarily alleviates the symptoms but the resulting
interference with sleep or sitting can be inconvenient and exhausting3. The condition is usually
persistent once established 4-6. The prevalence of the condition has been reported to be as great as
2.7% of the general population suffering moderate or severe symptoms 2 – 3 times per week 7-9 .
The clinical features of a symmetrical movement disorder with no local neurological abnormality has
suggested a central mechanism although a distinction from other such disorders is that the movement
is not totally involuntary: while the final form it takes is voluntary, the impulse to move is neither
desired nor deniable. Treatment has generally been by centrally-acting classes of drugs: dopamine
agonists eg. pergolide 10and ropinirole 11, benzodiazepines such as clonazepam, anticonvulsants or
opioids. Partial suppression of the symptoms can be achieved during treatment with relapse usual
when the drug is discontinued. Counterstrain is a manual treatment method discovered by Jones12,
a US osteopathic physician. The procedure involves locating the specific tender ‘Jones’ points in the
body, moving the body or limb into a position that reduces tenderness in the point, and maintaining
that position for ninety seconds before slowly returning the region to anatomical neutral. While Jones
used the treatment for musculoskeletal pain and impairment, Longden discovered that certain Jones
points on the pelvis appeared to be specific to RLS 12, and treatment of them could relieve RLS
symptoms. Longden’s technique was tested by Peters in a case series of twenty volunteer
participants with RLS13 and the findings suggested that a course of four treatments might provide
substantial symptom relief which may persist after completion of the course of treatment. A
randomized trial was therefore planned in order to investigate the short-term specific efficacy of this
method. The trial was titled CARL, Counterstrain Assessed for Restless Legs.

Methods
The study was designed as a pilot, single-blind, randomized controlled trial to test the specific efficacy
of Longden’s technique. Ethical approval for the protocol was obtained from the London Col ege of
Osteopathic Medicine Research Ethics Committee. The trial took place at the Osteopathic Association
Clinic in central London and adhered to the published protocol 14 which is outlined below.
Recruitment: Participants were volunteers recruited between October 2009 and December 2010
through advertisements on a RLS support group dedicated web-site and in a free local daily newspaper.
The eligibility criteria were a positive diagnosis on the International Restless Legs Syndrome Study
Group diagnostic criteria,15 aged 18 years and over; moderate to severe symptoms defined as a total
score of 14 or more on the International Restless Legs Scale (IRLS)16; and a consistent level of RLS
symptoms for at least 3 months.
The exclusion criteria were an age of less than 18 years; an IRLS score of less than 14; remission of
RLS in the past 3 months; the presence of contra-indications to the treatment such as hip pathology;
current use of drugs known to precipitate RLS such as antidepressants; or factors associated with
RLS such as uraemia or iron deficiency anaemia.
All those responding to advertisements were contacted and a preliminary screening for eligibility was
conducted. Those interested in participation and potentially eligible were sent the Patient Information
Sheet for the trial and invited for a clinical assessment in which they were screened fully for eligibility.
For eligible subjects, the implications of entry to the trial were discussed, any resulting questions fully
answered, and written consent requested. Those consenting were formally entered into the trial.
Randomization and masking: Sealed envelope randomization was used: a stack of forty sealed
envelopes contained equal numbers of A (active) or B (sham) allocations. The envelopes were
sequentially numbered on the outside with a study ID after being shuffled for twenty minutes to
ensure random distribution. After trial entry the next envelope in the series was drawn by the
researcher [RM] and opened in an adjacent room, the participants’s ID and treatment allocation were
recorded in the trial log and the treating clinician [TP] was informed of the allocation. Treatment
according to allocation was then carried out by the clinician who refrained from discussing the specific
purpose of each part of the procedure, in order to maintain blinding. The timing of appointments and
the waiting area arrangement precluded subjects discussing their treatments with each other. A short
post-treatment questionnaire was used to assess whether blinding was successful.
Intervention: All participants were offered four weekly sessions of manual treatment within nine
weeks of trial entry, carried out by one osteopathic physician [TP]. The control intervention (B)
involved Counterstrain applied to the lower half of the body treating any positive (tender) Jones points
found, but avoiding Longden’s RLS-specific points. The active intervention (A) was identical to the
control intervention, except that Longden’s RLS-specific points were treated, and this was reinforced
by a self-treatment procedure to the same points. The self-treatment was for home use and aimed to
duplicate the administered technique in that the patient monitored by palpation the tenderness of their
Longden’s points and reduced tenderness by positioning themselves. To maintain blinding, they were
simply told that this was an exercise often used for RLS.
Those taking drugs for RLS were told that should they experience a major reduction in symptoms
they could, in consultation with their clinician, progressively reduce their dosage and report this to the
researchers.
Assessment and outcome measures:
The primary outcome measure was the International Restless Legs Scale [IRLS] total score: a ten
question report of patient symptom severity, scored with increasing severity from 0 to 40. The
reliability of the IRLS had been tested 16 and found to be internally consistent, valid and reliable.
Secondary outcome measures were:
1. Sleep loss: patient reported average hours lost per night due to RLS in the last five days. 2. Patient Specific Outcome Score [PSOS]: a study-specific measure of improvement for which the patient nominated, at baseline, up to three goals for treatment and assigned weights to their importance; at six weeks after entry, linear analogue scales were used to capture their view of the degree to which they had achieved their goal(s), and a weighted score computed. 3. Global Improvement Scale [CGI-I]: a patient-completed six point scale of overall change 17.
The IRLS, sleep loss and drug usage were completed at base-line. Follow-up questionnaires of CGI-I
and sleep loss were completed at home five days after first attendance and then on the same day
each week for six weeks. These were held in a bound book that retained pressure sensitive
duplicates of the forms after the top copy was removed to be returned at the next clinic attendance or
by post (in reply-paid addressed envelopes). IRLS and PSOS were completed at week six only.
Questionnaires not received within a week of the expected date were followed up by telephone or
email to elicit any obstacles to compliance. The integrity of the patients’ scores was ensured not only
by secure storage of returned questionnaires at the research centre, but also by asking each patient
to retain safely the bound ‘questionnaire book’ containing the duplicate copies of their responses, and
to make it available only to an independent auditor of the research integrity.
Adverse effects were monitored at the weekly face-to-face contacts with the clinician and the
researcher, and by telephone follow-up at eight weeks after entry. No persisting side-effects of
Counterstrain had been previously reported to permit design of a structured record form.
Analysis:
The mean scores at each time point were computed for Group A and Group B, on an intention to treat
basis. The change in scores from baseline to six weeks after entry were computed for IRLS and sleep
loss, for each participant. The difference in mean scores between groups, with confidence intervals,
were then computed, and using analysis of variance methods, differences between the improvements
in group A compared with group B were compared (unpaired t tests, 2 tailed). The mean CGI-I score
at 6 weeks was compared between groups. The primary outcome measure was the mean change in
IRLS score from 0- 6 weeks. In the intention to treat analysis, the scores at week six were imputed if
a score was available for week 5. Otherwise, patients with a missing score were omitted from
summary statistics for that measure. A sensitivity analysis was conducted 18 using different more
conservative assumptions: if scores were missing then no change from baseline was assumed, and
all patients were included in the analysis.
Power Calculation: As this was a pilot trial, formal estimates of the power of the study were not made.
Results
The flow of patients into the trial is shown in Figure 1. The response to advertising the trial was brisk,
with 241 people responding to advertisements. Of these, 120 volunteered sufficient information by
telephone or email for the initial screening for eligibility, which found 64 (53%) potentially eligible.
They then attended and were assessed by the clinician who found 39 (32·5%) fulfilled the eligibility
criteria for the trial. They were formally invited and all 39 consented to enter the trial.
Twenty patients were randomized to the active intervention arm A and 19 patients to control arm B. The
baseline characteristics of the two groups were similar (see Table 1). Ages ranged from 24-76 years with
a mean of over 50 years. Sleep loss ranged from 0-5·5 hours. The IRLS score ranged from 16-35 with a
mean score of over 20. In Group A 63% of participants had a family history of RLS compared with 42% in
Group B. In Group B seven subjects were taking medication for their RLS: five ropinirole. Of the seven
only one reduced their dose: one using ropinirole from 1·5 to 0·5 mgs daily. In Group A, two subjects were
taking dopamine agonists and did not change dose during the study period.
Patients completed the questionnaires regularly and drop out was low (2 patients, 5% in total). In
group A, one patient (ID 32) withdrew before any treatment; one patient (ID 21)dropped out after five
weeks due to the onset of acute low back pain (of which she had a recurrent history); she had no six-
week scores. In group B, there were no dropouts during the study period of 0-6 weeks, but a few
patients failed to complete all the measures at 6 weeks, one patient (ID 15) had missing data for IRLS
and PSOS; two patients (ID 9 and 37) had missing scores for sleep loss.
Blinding was assessed immediately after the first treatment using a 100mm linear analogue scale with
one end representing total certainty of a Group A allocation and the other similar certainty of being in
Group B. Nineteen participants (50%) recorded a score of 50 (total uncertainty). The remaining 50%
participants had an impression of which group they were in, averaging 12mms from the midpoint, of
which 11 were correct and 8 incorrect. These results are consistent with guessing.
Table 2 shows the main results. The IRLS score decreased between baseline and week six by 10·33
points in group A, compared to 2·28 points in group B. This represented an improvement of 42·2% in
the active group compared to 8·7% in the controls. The difference between groups (the primary
outcome measure) of 8·06 points (95% CI 3·15 to 12·96, p=0·0021) was highly statistically significant
in favour of the active intervention. At six weeks, two of the secondary outcome measures also
showed significant differences between groups: the patient-rated improvement (CGI-I) was 0·84
points greater in group A by (95%CI 0·12 to 1·56) compared to group B, and the Patient specific
outcome score (PSOS) was 27·83 points higher in group A (95%CI 5·10 to 50·57). Sleep loss showed
no significant difference between groups. Drug usage was low among the participants, and was not
evaluated. The sensitivity analysis results were slightly less favourable to the active intervention but
the primary outcome measure remained statistically significant. The change in IRLS score showed a
difference between active and control arms of 7·14 (95%CI 2·39 to 11·89, p=0·0043). The PSOS
scores at six weeks remained significant different (p=0·041) but the CGI-I scores were non-
significantly different (p=0·096) between arms.
There were no adverse events reported from treatment; one patient dropped out at week five due to
recurrence of a back pain problem.

Discussion
Although, as a pilot trial, results should be viewed with caution until confirmed by larger definitive
studies, outcomes showed a highly significant short-term benefit from Counterstrain treatment of the
RLS-specific points identified by Longden. The trial suggests the Longden technique may have
specific efficacy, since the control intervention resembled the active intervention very closely, and
differed only in the use of the RLS-specific points. Blinding appeared effective and drop-out was low,
reducing the chance of bias. This is supported by the similarity of results from the sensitivity analysis.
The positive outcome of this trial of a manual treatment for RLS may stimulate debate about the aetiology and
patho-mechanics of RLS, currently thought to be a disorder of the central nervous system. Further research
into the mechanism of action of Counterstrain and the nature of the tender Jones points are also warranted.
Following a successful pilot, larger definitive studies should attract funding sufficient to allow group allocation
and data handling to be segregated from the clinicians managing the interventions and further assuring the
integrity of the data. It cannot be ignored that there are credibility issues when a strong treatment effect is
obtained by a method that must be implausible to neurologists and derives from a complementary therapy.
Such issues may impede the adoption of the method and even its evaluation by established centres.
In the intervention arm, Longden’s RLS-specific points were treated both by the osteopathic physician
and by the patient at home, therefore it is not possible to attribute the effect to one or both of these
interventions. Another study would be needed to test the effects of physician treatment alone or the
self-treatment regime alone. Due to small sample size, sub-group analyses were not possible. As the
intervention was carried out by one practitioner, the results may be different if more practitioners were
included. Adoption of the method generally would involve developing a training programme for
therapists: on the other hand demonstration that patient self-treatment can be effective should lead to
developing and evaluating a patient education programme.
This pilot trial has provided an estimate of parameters such as accrual and drop-out rates, which will
inform the design of future trials. Further research is needed to confirm this positive finding and to
investigate the duration of persistence of symptom relief, after the course of four treatments. If symptoms
are relieved for some months, say, then manual treatment could offer a cost-effective alternative to
lifelong medication (the annual drug cost being typically £400 per patient). For the 20% of patients who
experience troublesome side effects from the medication 20, such an option could be welcome.
A meta-analysis of 14 trials of dopamine agonist treatment of RLS, 19involving over 3,000 patients,
indicated a weighted mean difference (WMD) of -4·93 IRLS points (95% CI, – 6·42 to –3·43; P<.001)
between active and placebo groups. The difference of -8·06 IRLS points ( -3·15 to -12·96; p 0·0021 )
in the CARL trial suggests that manual therapy could be comparable in effect size.
RLS is quite common (1-2% of the population), and often persists lifelong, with considerable impact
on quality of life. This is the first reported trial of this manual technique for RLS. The results are
encouraging and merit further investigation. In contrast to the usual care of RLS with centrally acting
dopamine agonists, manual treatment has no side effects and, if relief of symptoms persists, could be
a cost-effective alternative option for patients.
Personnel : Treatment was given by Dr Theo Peters [TP] a registered medical practitioner and
osteopath who has been trained in the method developed by Dr Douglas Longden. Initial design of
the study was by Dr Roderic MacDonald [RM] a registered medical practitioner with full osteopathic
training. The analysis was planned and conducted by Dr Janine Leach, registered osteopath and
Senior Research Fellow at the University of Brighton. All authors took part in the preparation of this
report for publication and agreed its final form.
Funding: Clinic Facilities were provided by registered charities, Osteopathic Trusts and the College
of Osteopaths. The Association for Medical Osteopathy made a grant to cover stationery, postage,
travel and advertising. The clinicians were unpaid.
Acknowledgements : We are grateful for advice on trial design by Dr Damien Ridge,
Complementary and Alternative Medicine Reader in the School of Integrated Health, University of
Westminster and other members of their faculty. We are grateful to the Osteopathic Trusts and the
College of Osteopaths who provided clinic space and reception services without charge and the
Association for Medical Osteopathy who provided some funding.
Conflicts of Interest: The clinicians involved in the trial could expect, in the future, to provide
treatment and training involving the methods assessed should their worth be established.
1. Ekbom KA. Restless legs; a report of 70 new cases. Acta Med Scand Suppl. 1950; 246: 64-8.
2. Ekbom KA. Restless legs (letter). JAMA. 1946; 131 (5): 481.
3. Leschziner G, Gringras P. Restless legs syndrome. BMJ. 2012; 344: e3056.
4. Hening W, Allen R, Earley C, Kushida C, Picchietti D, Silber M. The treatment of restless legs syndrome and periodic
limb movement disorder. An American Academy of Sleep Medicine Review. Sleep. 1999; 22(7): 970-99.
5. Walters AS, Hickey K, Maltzman J, Verrico T, Joseph D, Hening W, et al. A questionnaire study of 138 patients with
restless legs syndrome: the 'Night-Walkers' survey. Neurology. 1996; 46(1): 92-5.
6. Earley CJ, Silber MH. Restless legs syndrome: understanding its consequences and the need for better treatment.
Sleep Med. 2010; 11(9): 807-15.
7. Innes KE, Selfe TK, Agarwal P. Prevalence of restless legs syndrome in North American and Western European
populations: a systematic review. Sleep Med. 2011; 12(7):623-34.
8. Pereira Junior JC, Silva Neto JL, Pradella-Hallinan M. Restless legs syndrome in subjects with a knee prosthesis:
evidence that symptoms are generated in the periphery. Clinics (Sao Paulo). 2011; 66(11): 1955-9.
9. Yeh P, Walters AS, Tsuang JW. Restless legs syndrome: a comprehensive overview on its epidemiology, risk factors, and treatment. Sleep Breath. 2011. 10. Trenkwalder C, Hundemer HP, Lledo A, Swieca J, Polo O, Wetter TC, et al. Efficacy of pergolide in treatment of
restless legs syndrome: the PEARLS Study. Neurology. 2004; 62(8): 1391-7.
11. Trenkwalder C, Garcia-Borreguero D, Montagna P, Lainey E, de Weerd AW, Tidswell P, et al. Ropinirole in the
treatment of restless legs syndrome: results from the TREAT RLS 1 study, a 12 week, randomised, placebo controlled
study in 10 European countries. J Neurol Neurosurg Psychiatry. 2004; 75(1): 92-7.
12. Jones LH. Spontaneous release by positioning. The DO. 1964; 4: 109 -16.
13. Peters T. Restless legs. Osteopathy Today. 2002; 8(8): 12-3.
14. MacDonald R, Peters T, Leach J. A randomised controlled trial of positional release manipulation (counterstrain) in
the treatment of restless legs syndrome. International Musculoskeletal Medicine. 2011; 33 (1): 2125.
15. Allen RP, Picchietti D, Hening WA, Trenkwalder C, Walters AS, Montplaisi J. Restless legs syndrome: diagnostic
criteria, special considerations, and epidemiology. A report from the restless legs syndrome diagnosis and
epidemiology workshop at the National Institutes of Health. Sleep Med. 2003; 4(2): 101-19.
16. Walters AS, LeBrocq C, Dhar A, Hening W, Rosen R, Allen RP, et al. Validation of the International Restless Legs
Syndrome Study Group rating scale for restless legs syndrome. Sleep Med. 2003; 4(2): 121-32.
17. Berk M, Ng F, Dodd S, Callaly T, Campbell S, Bernardo M, et al. The validity of the CGI severity and improvement
scales as measures of clinical effectiveness suitable for routine clinical use. J Eval Clin Pract. 2008; 14(6): 979-83.
18. Hollis S, Campbell F. What is meant by intention to treat analysis? Survey of published randomised controlled trials.
BMJ. 1999; 319(7211): 670-4.
19. Baker WL, White CM, Coleman CI. Effect of nonergot dopamine agonists on symptoms of restless legs syndrome.
Ann Fam Med. 2008; 6(3): 253-62.
20. Allen RP, Ondo WG, Ball E, Calloway MO, Manjunath R, Higbie RL, et al. Restless legs syndrome (RLS)
augmentation associated with dopamine agonist and levodopa usage in a community sample. Sleep Med. 2011; 12(5):
431-9.

This is a post-print version of an original article which has been peer reviewed and
accepted by International Musculoskeletal Medicine [ www.maney.co.uk ] and is now
in-press with a publication date in 2012 .
The protocol for this trial has been published and is accessible via
http://www.ingentaconnect.com/content/maney/imm/2011/00000033/00000001/art00005

Source: http://www.jiscs.com/PDFs/CounterstrainManipulation.RestlessLegSyndrom.McDonald2012.pdf