Abstract
Abstract
Background:
Phantom-limb syndrome can significantly impact on amputees' quality of life and their functional capacity. Conventional treatment approaches have a poor rate of success in alleviating symptoms. Acupuncture has been advocated as an effective alternative treatment; however, there has not been a systematic review of the evidence base for this.
Objective:
The aim of this review was
Methods:
This review comprised three phases: (1) an electronic database search to identify potential articles: (2) extraction of data from accepted articles; and (3) rating of internal validity and strength of the evidence provided in the accepted articles.
Outcome Measures:
An Effective Public Health Practice Project quality assessment tool (quantitative studies), a Critical Appraisal Skills Programme quality assessment tool (qualitative studies), and Agency for Health Care Policy and Research guidelines (strength of evidence) were used to assess the evidence.
Results:
Level C evidence showed acupuncture treatment reduced phantom-limb pain and sensation (14 studies), improved functional capacity or mobility (5 studies), and reduced levels of analgesic use (3 studies).
Conclusions:
Acupuncture therapy has been shown to have a positive effect on the symptoms of phantom-limb syndrome. However, further investigations of superior quality are needed to support these findings and determine the most effective method of acupuncture for this purpose.
Introduction
Several theories have been proposed regarding the mechanisms of phantom-limb syndrome, with central and peripheral changes in neural structure thought to be primarily responsible. 6 At a peripheral level, the severance of peripheral nerves during amputation and the formation of neuromas are thought to be responsible for symptoms, 7 while, at a central level, the cortical remapping that occurs when the area of the brain that controlled the limb before it was amputated no longer has a function is thought to contribute. 8 The neuromatrix theory proposes that conscious awareness and the perception of self are generated in the brain and modified throughout one's lifetime by various sensory inputs to create a neurosignature, which determines how a specific body part is consciously perceived. 2 The persistence or reorganization of the neurosignature after limb amputation is thought to be a cause of phantom-limb syndrome. 9 PLS is also often experienced by patients with congenital limb deficiency; however PLP, is rarely seen in this population, 10 and the mechanisms are not fully understood. 11
Phantom-limb syndrome is a complex condition, with physical, psychological, and perceptual aspects, making it a challenge to manage successfully. Conventional management—including pharmacology, nerve blocks, physical therapy and surgery—has provided limited help in alleviating phantom-limb syndrome. 3 Therefore, patients have turned to alternative therapy for management of symptoms. Acupuncture is a popular alternative therapy and has long been used as an intervention for phantom-limb syndrome. Acupuncture is a recommended treatment for this condition in well-established textbooks, 12 and at several prominent acupuncture courses. 13
Acupuncture is thought to stimulate the central nervous system to release neurotransmitters, hormones, or the body's natural pain-relieving endorphins, 14 and alters how blood pressure, blood flow, and body temperature are regulated and respond to pain. 15 To alleviate PLP, acupuncture needs to engage the nervous system to override the response to mismatched information. Acupuncture to the intact limb sends a normal afferent input to the nervous system, eliciting an analgesic effect. 16 Acupuncture stimulation of points in the ear, stump, scalp, and contralateral limb have been reported to help alleviate symptoms in subjects with phantom-limb syndrome.16,17
While acupuncture is frequently used and advocated in the treatment of phantom-limb syndrome and has been shown to have a beneficial effect, there has not been a systematic review of the evidence base for this. Therefore the primary aim of this systematic review was to determine the effectiveness of acupuncture as a treatment for phantom-limb syndrome, encompassing PLP and PLS. Secondary aims were to investigate the impact of acupuncture on functional limitations and analgesic usage and to investigate whether or not and where further research is needed.
Methodology
Overview
A systematic review of the literature was undertaken using the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) guidelines, 18 to determine the effectiveness of acupuncture for treating phantom-limb syndrome. This review comprised three phases: phase 1 involved a comprehensive database search; phase 2 comprised dual screening of studies by 2 independent reviewers (S.M.M. and G.A.K.); and phase 3 involved the use of validated tools to establish internal validity of included studies by 2 independent researchers (S.M.M. and G.A.K.), as well as quantifying the strength of evidence. Results were then categorized and discussed, using a thematic approach as described in a previous study. 19
Phase 1: Database Search
A list of key words was drawn up for the terms
Box 1: Search Terms
AND
Phase 2: Screening of Literature and Data Extraction
The titles, abstracts and keywords were reviewed by 2 independent assessors (S.M.M., G.A.K.) to determine if these items fulfilled predefined inclusion criteria. Inclusion criteria for this review were that studies had to assess subjects who had PLP or PLS post amputation. Amputation had to involve partial or full removal of one or more of the four limbs and congenital limb deficiency was not included, as the mechanisms for this condition are not understood fully. 11 Treatment had to include acupuncture needling (with or without laser and moxibustion) and the effects of treatment on symptoms, including pain, had to be assessed pre- and post-intervention. All types of studies were included, with no limitation on year. Only English-language studies were included.
The 2 researchers met to discuss which articles should be included in the final review, and a consensus was reached (Fig. 1). The full articles of the studies that had been thought suitable were then obtained, with data extracted using a detailed proforma to assess suitability for inclusion. These were again independently reviewed by 2 assessors (S.M.M., G.A.K.), with any studies that did not fulfill inclusion criteria being excluded. From this process, a final list of articles for review was assembled. As this review included studies of mixed methodologies, meta-analysis was not possible and effect size could not be examined. Results were examined using a narrative analysis, and grouped according to outcomes of interest.

Flowchart of study inclusion.
Phase 3: Quality Assessment and Strength of Evidence
A “gold standard” assessment tool to assess nonrandomized control trials (non-RCTs) is currently not available. Therefore, two appraisal tools were used to classify the internal validity and quality of the included studies. The Effective Public Health Practice Project (EPHPP) quality-assessment tool 21 was used to analyze quantitative studies, while the Critical Appraisal Skills Programme (CASP) quality assessment tool 22 was used to analyze qualitative studies. The internal validity and quality of the included studies was assessed independently by 2 researchers (S.M.M. and G.A.K.), with ratings discussed until a consensus was reached.
The content and construct validity and inter-rater reliability of the EPHPP have met acceptable standards. 23 This assessment tool has been identified as one of the most appropriate for assessing RCTs and non-RCTs. 24 It has previously been used in systematic reviews with similar methodologies to this one.25,26 The tool is used to rate six aspects of each study—(1) selection bias, (2) study design, (3) confounders, (4) blinding, (5) data collection methods, and (6) withdrawals/dropouts—as “weak,” “moderate,” or “strong.” Overall quality is classified as “strong” if all sections have strong ratings and no weak ratings; “moderate” if there are less than four “strong” ratings and one “weak” rating; and “weak” if there are two or more “weak” ratings.
The CASP tool 22 is used to assess qualitative research with ten questions that address the rigor of the research methods, the credibility of the findings, and the relevance of the findings. Each question requires a “yes” or “no” answer. The CASP has previously been used in systematic reviews with similar methodologies to the current review.27,28 Quality of a study is classified as “strong” if more than two-thirds of the answers are “yes”; moderate if between four and six “yes” answers are given; and weak if more than han two-thirds of the answers are “no.” 27
The strength of evidence for the current review was graded according to the Agency for Health Care Policy and Research (now known as the Agency for Healthcare Research and Quality) guidelines. 29 The original rating scale categorized evidence on a 4-point scale (A to D). 30 For the current review, a previously modified version was used that involved a 5-point scale. 27 With this scale, quality of evidence ranges from the highest quality (Level A) to the lowest quality (Level E).
Results
Literature Search
From a total of 6733 database results, sixteen studies fulfilled the inclusion criteria for the current review: ten single-case studies3,17,31–38; three cross-sectional studies39–41 ; two case-study series16,42; and one non-RCT 43 (Table 1). Two case-study series reported treating subjects who had a variety of conditions with acupuncture; in these cases, information was extracted in relation to the description of treatment of the subject with phantom-limb syndrome and, therefore, these two series were treated as single-case studies in the current review.3,35 Researchers in one study 17 performed one session of laser treatment without needling and one session of acupuncture needling; for the purpose of this review, only the needling treatment was included. In the three cross-sectional studies, information on the efficacy of acupuncture treatment has been extracted from a larger set of results with information from only the participants who underwent acupuncture therapy being used.
Intervention group as a whole prior to classification into 3 groups, according to chronicity, and control group.
Time units: x/24=number of hours; x/7=number of days; x/52/ number of weeks; x/12=number of months.
Meridians: LR, Liver; SP, Spleen; ST, Stomach; LI, Large Intestine; GB, Gall Bladder; HT, Heart; LU, Lung; PC, Pericardium; GV, Governor Vessel.
US, United States; yr(s), year (s); PLP, phantom-limb pain; VAS, visual analogue scale, Rx, treatment; min, minutes; ↓, reduced; Pt, patient; UK, United Kingdom; PLS, phantom-limb sensation; RTA, road-traffic accident; NRS=numerical rating scale; P&Ns=pins and needles; ECs; elbow crutches; mo(s), month(s); ↑, increased; RCT, randomized controlled trial; PENS, percutaneous electrical nerve stimulation; TENS, transcutaneous electrical nerve stimulation.
Quality of Evidence
Eleven of the included studies were classified as quantitative in nature, while five were qualitative. Studies that combined the two types of methodology (i.e., quantitative and qualitative) were considered as quantitative for this review, given that the primary outcome measure in each case was quantitative. This was with the exception of one study, 31 which did not provide enough information for the study to be classed as quantitative (the quantitative measure was performed pre- but not post-intervention). Of the quantitative studies, all eleven were assessed as being of weak quality when analyzed using the quality EPHPP assessment tool (Table 2). Of the qualitative studies, four were assessed to be of weak quality,3,31,34,38 and one was categorized as having strong quality, 41 using the CASP quality assessment tool (Table 3).
S, strong; M, moderate; W, weak.
Six studies dealt with upper-limb amputees only.31,33–36,38 Six studies also dealt exclusively with lower-limb amputees.3,16,32,37,39,42 Three studies40,41,43 dealt with a mixture of upper- and lower-limb amputees, while one study 17 involved treating a subject with bilateral upper- and lower-limb amputations. Given that one study 40 surveyed health care providers instead of patients; no information was available on types of amputations included in that study and, so that study is absent from the following listings. Sites of amputations seen were transtibial,16,36,37,42 knee disarticulation, 36 transfemoral,16,17,36,42 hemipelvectomy,16,36 transpelvic, 32 hip disarticulation,17,36 two thirds of thumb, 34 four digits of hand, 35 wrist disarticulation, 36 transradial,17,36,38 transhumeral,17,36,43 shoulder disarticulation33,36 and interscapulothoracic. 43 Exact sites of amputation were not stated by three studies.3,31,41 In one study, 39 the researchers stated that the most common amputation seen was transtibial, but they did not elaborate on the range of amputations.
Amputations were classified as traumatic if the initial injury was traumatic, regardless of subsequent revisions. Nine studies had subjects with exclusively traumatic injuries.3,17,31,33–38 One study considered exclusively medical amputations. 32 Four studies had subjects with a mixture of traumatic and medical amputations.16,39,41,42 The researchers of one study 43 stated that the majority of amputations were needed because the patients had sustained industrial accidents and road-traffic accidents (RTAs); however it was not stated whether medical amputations were also included. The most common cause of amputation was an industrial accident.31,35,36,38,42,43 There were also amputations required because of cancer,16,32,42 military injuries,3,17,41,42 RTAs,16,33,43 boating accidents, 37 embolisms, 42 falls from a height, 42 and congenital talipes. 16 The researchers of two studies stated only the most common causes among their subjects: trauma; industrial accidents; and RTAs.39,43 Sherman at al. 41 found that the majority of their respondents had military-related amputations. The remaining respondents were described as having amputations because of disease, but types of diseases were not specified. Leung and Spoerel 34 reported their participant's amputation as traumatic; however, the exact cause of this amputation was not stated clearly.
Time Since Amputation
Four studies dealt with subjects in the acute stage (i.e., within 1 month of amputation).32,36, 37,43 Three studies included subjects in the subacute stage (i.e., between 1 and 6 months of amputation.16,17,43 Eight studies included patients with chronic symptoms (i.e., ≥6 months.16,33–35,38–40,42 The length of time since amputation was not documented by two studies.31,34 Hao and Hao 3 stated that it was several months since their participant's amputation but did not state how long the participant had the symptoms. Sherman et al. 41 stated that the average number of years since amputation was 26; however, the range was not reported and it was stated that many respondents had had amputation in the last year or so.
Previous Treatment
It was not possible to extract information about previous treatment from the three cross-sectional studies.39–41 The researchers of four studies did not state whether their subjects had received other treatments prior to acupuncture therapy.35–37,42 The researchers of the remaining nine studies described various treatment modalities that had failed to alleviate symptoms prior to acupuncture intervention, including such treatments as pharmacological management, nerve blocks, physical therapy, and neuromal excisions. Eight studies mentioned unsuccessful pharmacological management.3,16,17,31–33,38,43 Researchers of four studies described subjects receiving nerve blocks that did not alleviate symptoms.31,33,34,38 Three studies found that physical therapy did not relieve symptoms.17,31,43 Two studies reported unsuccessful neuromal excisions.34,38 Other unsuccessful interventions included a dorsal-column stimulator, 16 massage, 17 hypnotherapy, 38 transcutaneous electrical nerve stimulation (TENS), ultrasound, and dorsal-root entry lesion. 33
Number of Treatment Sessions with Acupuncture
Five studies involved treating 1 or all subjects for one treatment session using acupuncture.3,16,17,32,34 Three studies involved treated subjects for four sessions with acupuncture.16,33,37 Other studies involved completing six 35 and seven 36 sessions of acupuncture. Aldrete and Ghaly 31 treated a subject over a 6-month period; however, the number of sessions applied was not stated. Similarly Monga and Jaksic 38 treated a subject with acupuncture over a 3-month period but did not state the number of sessions that were applied. Liaw et al. 43 treated subjects for between one and seven sessions; some subjects discontinued treatment when they were satisfied with the level of improvement experienced. Xing 42 treated patients once per day for 3–4 weeks (twenty-one to twenty-eight sessions). It was not possible to extract data about the number of treatment sessions from the three cross-sectional studies.39–41
Sites of Acupuncture Treatment
Researchers of five studies used points on the contralateral intact limb corresponding to sites of PLP or PLS on the absent limb.16,31,36,37,43 Of these five studies, only Bradbrook 16 and Liaw et al. 36 used recognized acupuncture points. Lu 37 treated a subject with percutaneous electrical nerve stimulation at the thoracic, lumbar, and sacral spine, bilateral knees and the contralateral intact foot. Johnson et al. 33 used a specific point on the cervical spine, which was found to relieve symptoms; however, this was not a recognized acupuncture point. Monga and Jaksic 38 used Large Intestine, Pericardium, Lung, and Heart points on the contralateral intact limb to treat a transradial amputation and Large Intestine points on the affected side; however, these researchers did not justify their selection of points. Xing 42 treated a series of transfemoral and transtibial amputees using Yaojiaji, Gall Bladder, and Bladder points on the affected side; Gall Bladder, Spleen, Stomach, and Liver points on the contralateral intact limb; bilateral Pericardium, Heart, Gall Bladder, and Governing Vessel points, along with the sensory area of the scalp; however this selection of points was not justified. Researchers of three studies16,32,36 chose points on the basis of their analgesic properties. Fulton 32 and Liaw et al. 36 also chose points for their relaxation-inducing properties. Scalp acupuncture was used by Hao and Hao 3 ; however, exact points were not stated. Auricular acupuncture was utilized by Jacobs and Niemtzow, 17 who placed one needle at the ipsilateral cingulate gyrus, and by Leung and Spoerel, 34 who inserted a needle into the ipsilateral locus point corresponding to thumb pain. Levine et al. 35 did not state what points were used. Levine et al. 35 and Xing 42 also used moxibustion in their treatments. Three studies35,37,42 reported the use of electroacupuncture. Monga and Jaksic 38 manually stimulated needles. Bradbrook 16 stated that the needles were stimulated but did not state how this was accomplished. Details of acupuncture treatment were not documented in the three cross-sectional studies.39–41
Long-Term Follow-Up
Six of sixteen studies followed up with subjects after treatment had finished. Aldrete and Ghaly 31 reported that, 18 months after treatment, their subject was pain free and receiving acupuncture for occasional aggravations. Bradbrook 16 stated that two of three participants remained symptom-free for the remainder of prosthetic rehabilitation. Johnson et al. 33 reported that their subject was still self-administering acupuncture with pain relief lasting only while the needles were in situ. Leung and Spoerel 34 described their subject at a 3-month follow up as continuing to have good hand function, despite some stump sensitivity. Lu's 37 subject was reported as having returned to work full-time on crutches and that this subject resumed an almost normal physical and mental life. Monga and Jaksic 38 stated that their subject was using TENS once per week to control pain (TENS was used to replace acupuncture so that subject could self-administer treatment) and no longer required pain medication.
Outcome Measures
All sixteen studies used subjective measures, while seven16,31,32,34,37,38,40 used a mixture of subjective and objective measures. All sixteen studies used subjects' self-reporting of symptoms. Four studies used a visual analogue scale (VAS).16,31,33,36 However Aldrete and Ghaly 31 only used the measure before treatment and not afterward. Fulton 32 used a numerical rating scale (NRS). Five studies used qualitative descriptors,3,16,32,34,37 for example, “telescoping” (the feeling of absent limbs retracting into the body), reduced feelings of heaviness, increasing feelings of warmth, cessation of entire pain syndrome, and normalization of sensation. Four studies31,33,35,38 used duration of pain relief after treatment as an outcome measure. Hanley et al. 39 used a 5-point anchored scale of treatment success/helpfulness ranging from 1 (“not at all helpful”) to 5 (“extremely helpful”). Sherman et al. 40 measured rate of success on an 11-point anchored scale of 0–10, ranging from 0 (“no success”) to 10 (“entirely successful”) and also a score generated by formula (sum of respondents calculated successes divided by number of respondents using treatment). Sherman et al. 41 used a qualitative scale ranging from “no effect” to “cure.” Jacobs and Niemtzow 17 used a 0–10 intensity scale. Levine et al. 35 asked subjects to rate percentage of pain (0%=“no pain” to 100%=“worst pain ever experienced”). Liaw et al. 43 used a verbal numerical scale of 0–10 (0=“no pain” to 10 “pre-acupuncture PLP level”). Two studies37,42 used percentage of pain resolution. Objective measures were not used to measure pain directly but were used to concentrate on changes in functional capacity and analgesic usage. Five studies used functional measures (e.g., ability to tolerate prosthesis),16,32,34,37,38 while three studies analyzed the impact on analgesic usage.31,32,38
Effectiveness of Acupuncture Treatment for Phantom-Limb Syndrome
Results were reported on using three common categories: (1) impact of acupuncture on PLP and/or PLS; (2) impact of acupuncture on functional capacity or mobility; and (3) impact of acupuncture on use of analgesics. All sixteen studies looked at the impact of acupuncture on symptoms of PLP and/or PLS. Five studies looked at the impact on subjects' functional capacity or mobility.16,32,34,37,38 Three studies looked at the impact of the intervention on analgesic use.31,32,38 Treatment was shown to be effective in patients with acute (<6 months, n=616,17,32,36,37,43) and chronic (≥6 months, n=716,33,34,38,40–42) symptoms (see Box 2).
Box 2. Summary of Results
• Acupuncture treatment effective in patient with acute (<6 months, n=6) and chronic (≥6 months, n=7) symptoms.
• Level C evidence that acupuncture treatment reduced phantom-limb pain and sensation (n=14).
• Level C evidence that acupuncture treatment improved functional capacity or mobility (n=5).
• Level C evidence that acupuncture treatment reduced levels of analgesic use (n=3).
Impact of Acupuncture on PLP and/or PLS
Consistent Level C evidence showed that acupuncture treatment reduced PLP and PLS (n=14; see Box 2). All sixteen studies looked at the impact of acupuncture on the symptoms of PLP and/or PLS. Fourteen of sixteen studies demonstrated that acupuncture was an effective treatment for phantom-limb syndrome (Table 1). Thirteen of these studies were of “weak” quality and one 41 was of “strong” quality. Improvements were shown in patients with acute, subacute, and chronic symptoms. Two studies of “weak” quality did not find that acupuncture had a positive effect on symptoms.35,39
All single-case studies, except for Levine et al., 35 showed positive results with acupuncture treatment. Symptoms were completely alleviated with acupuncture therapy in four of these studies.3,31,32,34 Aldrete and Ghaly 31 reported their subject as being pain-free and managing occasional aggravations with acupuncture at an 18-month follow-up, while Fulton 32 stated that another patient's symptoms disappeared with acupuncture needles in situ and that this relief lasted for 3 hours. Leung and Spoerel 34 reported that instant cessation of the entire pain syndrome was experienced with acupuncture therapy, with no phantom-limb reoccurrence at a 3-month follow-up.
Symptoms of phantom-limb syndrome were reduced in five studies.17,33,36–38 Jacobs and Niemtzow 17 reported pain relief still present at follow-up 1 day later, Monga and Jaksic 38 were able to provide 4–6 hours of symptom relief, while Johnson et al. 33 stated that analgesia only lasted while acupuncture needles were in situ. Levine et al. 35 described short-term relief of 50% of average pain with acupuncture. No increased degree or duration of relief was experienced with an increased number of treatment, and therapy was discontinued after the sixth session.
Of the three-cross sectional studies, two found that acupuncture therapy had a positive effect on phantom-limb syndrome. Sherman et al. 40 surveyed physicians' ratings of the success of various treatment options for phantom-limb syndrome they had encountered in practice, with acupuncture achieving one of the highest success scores. However, Hanley et al. 39 found that 67% of subjects (n=2) gave acupuncture a rating of 1 (“not at all helpful”) while 33% (n=1) gave it a rating of 3 on a scale of 1 (“not at all helpful”) to 5 (“extremely helpful”).
In both case-study series, all except 1 subject experienced relief of symptoms with acupuncture therapy. Two of 3 subjects in the study by Bradbrook 16 had their symptoms completely alleviated by acupuncture therapy. Xing 42 reported that 8 of 9 subjects showed an improvement in symptoms with acupuncture therapy. Liaw et al., 43 conducted a non-RCT of acupuncture and PLP. Acupuncture produced a statistically significant analgesic effect, especially following the first treatment or in patients who had received acupuncture within 1 week after the onset of PLP.
Impact of Acupuncture on Functional Capacity or Mobility
Consistent Level C evidence showed acupuncture treatment improved functional capacity or mobility (n=5; see Box 2). Five studies of “weak” quality assessed the impact of acupuncture on functional capacity or mobility.16,32,34,37,38 All five found that acupuncture therapy had a positive effect on subjects' function. Bradbrook 16 found that two of three participants were able to continue prosthetic rehabilitation while remaining symptom-free. Fulton 32 stated that one session of acupuncture therapy alleviated phantom-limb symptoms, which enabled a subject to mobilize with elbow crutches. Leung and Spoerel 34 described a subject whose hand had been “unusable” regaining functional use after a course of acupuncture therapy. Lu 37 reported on a subject returning to work full-time and resuming an almost normal physical and mental life post acupuncture therapy. Monga and Jaksic 38 stated that a subject was able to tolerate wearing a prosthesis, which was not possible prior to acupuncture therapy.
Impact of Acupuncture on Use of Pharmacological Analgesics
Consistent Level C evidence showed that acupuncture treatment reduced levels of analgesic use (n=3; see Box 2). Three studies of weak quality looked at the impact of acupuncture therapy on the amount of analgesic use by subjects.31,32,38 In all three cases, subjects were able to reduce the level of analgesic therapy post acupuncture intervention.
Discussion
Synopsis of Results
This review examined the effectiveness of acupuncture as a treatment for phantom-limb syndrome. Results were based on sixteen studies that included both quantitative and qualitative methodologies. Overall, there was notable evidence from fourteen studies that acupuncture reduces symptoms of PLP and PLS in phantom-limb syndrome. Single-case studies, case-study series, cross-sectional studies, and a non-RCT have used greatly differing forms of acupuncture therapy on subjects of different ages, amputation types, and levels of chronicity, and have shown positive results.
Impact of Acupuncture on PLP and/or PLS
Fourteen of sixteen studies demonstrated that acupuncture reduced PLP and/or PLS. This is consistent with recent research that has shown acupuncture to be an effective treatment for neuropathic pain, 44 arthritic pain, 45 chronic pain, 46 and migraine. 47 Treatment was shown to be effective in patients with acute16,17,32,36,37,43 and chronic16,33,34,38,40–42 symptoms, although it was shown to be more effective within 1 week of the onset of symptoms. 43 Benefits of acupuncture treatment were short-term in many cases. Similar to present findings in phantom-limb syndrome, a Cochrane review found that, for chronic low-back pain, acupuncture is effective for pain relief and functional improvement immediately after treatment and in the short-term only. 48 This may however be the result of inadequate doses of acupuncture being provided in some studies because of a lack of clear guidelines in this area. 49
Impact of Acupuncture on Functional Capacity and Mobility
Five studies showed that acupuncture had a positive effect on functional aspects of phantom-limb syndrome, such as ability to tolerate prostheses and mobilize.16,32,34,37,38 Acupuncture therapy has provoked feelings of “telescoping” (the perception that the phantom limb is progressively shortening in a proximal direction), eventually resolving up to the level of amputation. 50 This enabled tolerance of prosthetic limbs and thus functional capacity. 16 Acupuncture has previously been shown to improve mobility as well as reducing fatigue, depression, and sleeplessness in subjects with rheumatoid arthritis. 51 Acupuncture therapy has also been effective for improving function in subjects with chronic lower-back pain (cLBP)52,53 and stroke. 54
Impact of Acupuncture on Pharmacological Analgesic Intake
A Cochrane systematic review has suggested that acupuncture is possibly more effective than prophylactic drug treatment in providing analgesia for migraine headaches and has fewer adverse effects. 55 Acupuncture has been shown to be an alternative to pharmacological management for phantom-limb syndrome, without the associated side-effects of pharmacological agents, such as nausea and emesis 31 as well as memory impairment. 33 Some forms of pharmacological management control only PLP and not PLS, which is equally functionally restrictive, 32 while acupuncture targets both aspects of phantom-limb syndrome. Acupuncture has previously been shown to reduce levels of pharmacological analgesic use in patients with postoperative pain 56 and cLBP. 57
Study Type and Quality of Evidence
The type of studies conducted is a serious limitation in this area of research with the presence of ten single case studies and two case study series in this review. A published hierarchy of evidence suggests that the least likely studies to produce good evidence for practice are single-case studies. 58 The objective of a case study is to utilize recorded observations and narrative descriptions of an individual's characteristics and responses to treatment to provide a basis for making plausible inferences and generating hypotheses for future research.59,60 There are advantages to the case-study design in that it offers a rationale for the use of specific rehabilitation interventions or unique applications of treatment strategies and may offer suggestions for treatment modification or more efficient approaches. 61 Because of its exploratory emphasis, the case study is suitable for generating hypotheses. However, while a case study may include a report of objective findings, it does not provide very strong evidence in support of rehabilitation interventions because of the lack of controlled comparisons (e.g., between treatment and no-treatment conditions), making it difficult to generalize the results to other patients. 61 Yet, there is no doubt that case reports can provide useful information for further case series, reviews, and original research studies. 62
The quality of studies in this area is another serious limitation, with fifteen of sixteen rated as “weak.” Therefore, the evidence can only be categorized as Level C; although the evidence is generally consistent, it arises from poor-quality studies. Most studies were poorly controlled with significant risks of bias, with data analysis and examination of bias being very poor throughout. Although an acupuncturist administering a real or sham treatment cannot be blinded to the study condition, the potential for bias could be minimized if the acupuncturist were to have the least possible communication with each patient and if the evaluation were to be performed by an independent party. 63 Reporting quality was poor throughout many of the case studies, often with a deficiency of information on selection of subjects, potential influence of concurrent treatments, interventions performed, outcome measures, and follow-ups post treatment. These findings concur with research concerning the quality of acupuncture research, 64 which found that quality in reporting details of acupuncture interventions had not improved following the introduction of recommendations specific to this. 65
Types of Amputation, Time Since Amputation, and Previous Treatment
Reporting on the types of amputation and times since amputation was not adequate in some studies, limiting the value of their results in the context of current research. However, acupuncture therapy was shown to be effective in both upper- and lower-limb amputations, and in traumatic and medical amputations. Acute and chronic symptoms were treated successfully with acupuncture therapy, with almost all studies featuring subjects who had undergone unsuccessful treatment prior to acupuncture therapy. This may have had an impact upon the success of acupuncture therapy. The timing of treatment for musculoskeletal pain has been reported to be crucial, with early intervention often preventing chronic problems 66 and, therefore, it may be important not to wait for conventional treatments to fail before attempting acupuncture therapy. It was not explicitly stated in most studies if treatment was administered by a trained medical professional. It has been previously stated that reporting of the qualifications and experience of practitioners involved in acupuncture case reports is notoriously poor. 67 Guidelines have been formulated in order to improve reporting in acupuncture case reports 68 and should be followed.
Number of Treatment Sessions
None of the studies contained a plan for the number of treatment sessions prior to applying an intervention. Even within the non-RCT 43 and the two case-study series,16,42 subjects received differing numbers of treatments. Standardization in intervention is a central component of experimental research so that there is uniform manipulation of the independent variable. 69 While interventions will be individualized in practice, further research is essential to formulate guidelines for the number of treatment sessions required.
Sites of Acupuncture Treatment
There is a lack of consensus on appropriate acupuncture treatment sites for phantom-limb syndrome, and a lack of agreement on the optimal acupuncture treatment for any particular condition may mean that some patients do not receive the best treatment. 49 Sites of acupuncture were poorly reported or not justified in some studies.3,38,42 This is consistent with evidence that has identified problems in reporting quality of acupuncture case studies. 67 The researcher in one study stated that needles were stimulated but did not state the method used to accomplish this. 16 Many studies did not use recognized acupuncture points, which have been shown to have a superior analgesic effect compared to non-recognized acupuncture points. 70 However, these differences are relatively modest, suggesting that factors independent of effects of needling at specific points are important contributors to the therapeutic effects of acupuncture. 46 While both recognized acupuncture points and non-recognized acupuncture points been shown to have analgesic effects, further research should compare the two approaches in the context of phantom-limb syndrome to determine which is more effective.
Long-Term Follow-Up
Only six of sixteen studies performed follow-ups with subjects.16,31,33,34,37,38 In some studies, researchers treated subjects with only one session and did not follow-up beyond 1 day of intervention.3,17,32 Long-term follow up is essential to determine the beneficial or adverse effects of treatment over time. 71 Therefore, it is difficult to form a conclusion on the long-term effects of acupuncture therapy.
Outcome Measures
Most studies in this review focused primarily on pain intensity. While measuring pain intensity is an important part of assessment and helps evaluate the effectiveness of treatments for phantom-limb syndrome, using this one measure may fail to identify other factors, such as reduced quality of sleep, function, and depression. 72 As phantom-limb syndrome is a complex condition, researchers dealing with this problem should consider a multifaceted assessment of symptoms, using published guidelines for core outcome measures in the pain assessment area. 73 There are seven core domains recommended by the Initiative on Methods, Measurement, and Pain Assessment in Clinical Trials for assessment in studies of chronic pain, including pain, physical functioning, and emotional functioning. 73
It is crucial to include a mixture of both subjective and objective outcome measures in the assessment of phantom-limb syndrome as they are designed to measure different aspects of the same variable and give two distinct types of information. 74 Nine studies included in this review relied on subjective self-reported outcome measures only, while seven16,31,32,34,37,38,40 used a mixture of subjective and objective measures. Objective measures focused on changes in functional capacity and analgesic usage, indirect measures were focused on the pain experience. Given the complex interaction of sensory, affective, and cognitive components of pain, it is difficult for an objective measure to capture the experience of pain in a clinically useful way. 75 Physical functioning and performance tests are frequently used as a proxy for objective pain assessment. 76 Measures of physical function described by researchers in the studies examined in this review tended to be vague and based on subjects' self-reporting rather than on clinical assessments. For example, Leung and Spoerel 34 stated that their subject “continues to use her hand quite well.” It has been shown that there are only moderate correlations between self-reported activity limitation and corresponding clinician-measured performance tests. 77 The Functional Measure for Amputees measures function of lower-limb amputees in terms of prosthetic usage and is recommended for use in routine clinical practice. 78 Medication use and return to work were also used as objective measures by studies included in this review31,32,37,38 and have previously been used as objective outcome measures in a chronic-pain setting. 79
The use of subjective measures is justifiable as patients' self-reporting is the “gold standard” of pain assessment. 80 However, in this review just five studies used subjective outcome measures that have been shown to be valid and reliable,16,31–33,36 and these outcome measures were not always completed both before and after the interventions.31,36 Similarly, a review 81 of subjects with chronic pain or fatigue reported that most of the subjective methods featured had problems with reliability and/or validity. The VAS and NRS have been shown to be valid and reliable for assessing pain, 82 while the VAS has been reported to be reliable and repeatable for measuring phantom-limb phenomena and changes following acupuncture. 16 There is no outcome measure specific to PLP, but, as it is commonly classified as neuropathic pain, 1 validated tools designed to assess neuropathic pain could be utilized.83,84
Five studies included in this review used qualitative descriptors,3,16,32,34,37 giving insights into patients' individual experiences of illness, of which a good understanding is essential for providing care. 85 The McGill pain scale 86 is a valid and reliable assessment tool that incorporates these qualitative descriptors and assigns a numerical score that can be used to determine effectiveness of interventions. It would, therefore, provide superior-quality evidence in studies like these rather than the use of descriptors alone. Emotional functioning was not addressed in any of the included studies; but this parameter could be assessed with the Beck Depression Inventory 87 or the Profile of Mood States. 88 Overall, there needs to be increased utilization of valid and reliable measures rather than formulating unreliable scales for the purpose of individual studies.
Ethics
There was no mention of ethical considerations in any of the studies. Ethics help to govern the conduct of practitioners by coordinating their actions and also apply to the aims or goals of research. 89 Johnson et al. 33 reported using the VAS with a patient post brain injury who had from memory impairment and was later discharged with instructions to self-administer acupuncture. A study on use of the VAS with patients who have Alzheimer's disease (and therefore memory impairment) reported that the majority of midstage patients did not understood the purpose of these scales and recommended the continuation of the search for other tools to assess pain affect in this population. 90 Informed consent is one of the most important components of ethical experimentation; however, obtained consent has little meaning unless the subject (or guardian) is capable of understanding what is to be undertaken and all potential hazards. 91
Limitations
This review itself is not without limitations. Analysis of the most effective type of acupuncture intervention was limited because of heterogeneity among patient populations, interventions, and outcome measures. Selection of acupuncture points is often individualized among patients according to their signs and symptoms, 3 making good-quality RCTs difficult to obtain. Owing to the topic under review, observational, quantitative, and qualitative studies were included, as these comprise much of the information regarding acupuncture and phantom-limb syndrome. This made it necessary to adapt a discursive and thematic methodological approach, as meta-analysis and effect sizes can only be analyzed when only RCTs are included. This narrative approach has, however, been used in several reviews of acupuncture therapy.49,92 It has also been suggested that the inclusion of qualitative research within systematic reviews may provide a more complete reflection of the experience of subjects, and therefore, lead to more appropriate and effective management. 19
Another limitation of this review is the lack of an appropriate quality-assessment tool for single-case studies and case-study series. Certain sections of the EPHPP 21 and CASP 22 concerning groups, blinding, and randomization were not applicable to cross-sectional studies or case studies and, therefore, automatically generated a “weak” rating. Atkins, and Sampson 93 have formulated a checklist for quality assessment of single-case studies from which a valid and reliable quality-assessment tool could be developed. The inclusion of only English-language articles also limits the scope of this review, as many studies investigating acupuncture are in other languages (e.g., Chinese and Russian).
Implications for Practice and Areas for Future Research
The implications for practice are that practitioners treating patients with phantom-limb syndrome should be aware of acupuncture as a treatment modality and should also be aware that, while it is successful in patients with both chronic and acute symptoms, it is most effective within 1 week of onset 43 and should be not looked on as a last resort when conventional treatment fails.
Further better-quality research must be carried out to determine the form of acupuncture that is most effective, along with optimal treatment times and sites. Quality RCTs using a combination of validated reliable objective and subjective outcome measures are essential to provide evidence for the use of acupuncture therapy in this area. This is in line with the findings of Birch et al. 94 who reported that all examined reviews agreed that the methodological rigor of acupuncture clinical trials has generally been poor and that higher-quality clinical trials are necessary. This call to action from Birch et al. 94 in 2004 appears to have been largely unheeded in the context of research into acupuncture and phantom-limb syndrome. Greater adherence to STRICTA guidelines 64 is essential to improve the standard of research in acupuncture and to develop a mechanism in which guidelines for best practice with regard to the management of phantom-limb syndrome can be documented and shared.
It is also essential to examine the long-term effects of the intervention, as most studies have focused only on the short-term resolution of symptoms. To facilitate clinical and policy decision making, evidence on the long-term consequences of the outcomes used in trials is necessary. 95
Conclusions
There are reports of phantom-limb syndrome dating back to 1551. 2 As a condition that has been prevalent for a prolonged period of time, it is essential that an evidence-based framework for treatment be established. Acupuncture therapy has been shown to have a positive effect on the symptoms of phantom-limb syndrome in terms of pain, sensation, function, and analgesic intake. However, the majority of the literature is of weak quality and comprises single-case studies. Further investigations of superior quality are needed to enable health care providers to decide if acupuncture is an appropriate modality for use in the treatment of individual patients with phantom-limb syndrome and to determine the most effective method of acpuncture for this purpose.
Footnotes
Acknowledgments
The authors thank Ms. Avril Patterson, a Medical Librarian, at the Health Sciences Centre, University College Dublin, in Belfield, Dublin, Ireland.
Disclosure Statement
The authors declare no conflict of interest or competing financial interests exist.
