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Publicly Available Published by De Gruyter February 1, 2022

Adults with unilateral lower-limb amputation: greater spatial extent of pain is associated with worse adjustment, greater activity restrictions, and less prosthesis satisfaction

  • Jaclyn Megan Sions EMAIL logo , Emma Haldane Beisheim-Ryan , Ryan Todd Pohlig and Mayank Seth

Abstract

Objectives

This study’s primary purpose was to determine if the extent of bodily pain, as evaluated with pain body diagrams, is associated with prosthetic-related activity restrictions, adjustment, and satisfaction among adults with a major unilateral lower-limb amputation. A secondary objective was to evaluate between-days, test-retest reliability for pain body diagrams among adults with lower-limb amputation.

Methods

Adults with a lower-limb amputation that occurred ≥1 year prior participated in an online, cross-sectional research study. Outcome measures included pain body diagrams and the Trinity Amputation and Prosthesis Experience Scales-Revised, which evaluates post-amputation activity restrictions, psychosocial adjustment, and prosthesis satisfaction. Linear regression modeling was used to evaluate associations between the number of painful body regions and prosthetic outcomes, after considering covariates (alpha ≤ 0.010). A subset of participants recompleted pain body diagrams to evaluate between-days, test-retest reliability.

Results

Data from 74 participants (n = 32 female; n = 42 transtibial-level; n = 27 traumatic etiology) were available. Beyond covariates (i.e., age, sex, amputation level), the total number of painful body regions was significantly associated with all Trinity Amputation and Prosthesis Experience Scales-Revised subscales (p < 0.001–0.006), with the exception of Social Adjustment (p = 0.764). The total number of painful body regions explained 14.5, 11.8, 11.6, and 7.4% of the variance in Functional Satisfaction with the Prosthesis, Adjustment to Limitation, General Adjustment, and Activity Restriction, respectively. In a subset (n = 54), test-retest reliability for total number of painful body regions per body diagrams was good [intraclass correlation coefficient (ICC)3,1 = 0.84].

Conclusions

A greater number of painful body regions is associated with greater activity restriction, worse adjustment, and lower prosthesis satisfaction, supporting the need to enhance post-amputation pain management and both amputated- and secondary-site pain prevention.

Ethical committee number

IRB #1611862.

Introduction

Multisite pain, i.e., pain in two or more locations [1], has been associated with reduced health, lower quality-of-life, and increased healthcare utilization among individuals with peripheral joint pain [2]. Multisite pain is common after unilateral lower-limb amputation (LLA), with an estimated point-prevalence of 72.0 and 54.7% among females and males, respectively [3]. Post-amputation pain affects both the amputated region, resulting in phantom limb pain (PLP; pain in the amputated limb area) and residual limb pain (RLP; pain in the remaining part of the amputated limb), and secondary sites (including the low back and contralateral limb) [3]. Secondary-site pain may be attributed to poor prosthetic fit and/or alignment, postural changes, overuse, and deconditioning [4]. Greater RLP intensity has been associated with reduced prosthesis use [5]. Secondary-site pain can negatively impact physical function [6], and in some cases, may be of greater intensity than pain in the amputated limb [5]. Further, in an international study evaluating problems post-amputation, ‘sensations of pain’ was the most frequently reported category under the body structure and function domain (per the International Classification of Functioning, Disability, and Health) [7].

In clinical practice, pain intensity ratings are used to evaluate treatment efficacy. RLP and PLP, however, are rarely constant, but rather episodic with short durations [8]. As pain intensity ratings fail to capture pain duration, frequency, burden, and spatial extent, other pain-related metrics should supplement intensity. This is particularly relevant since a cohort study of 302 adults post-LLA found pain intensity was unrelated to outcomes [9], despite evidence of high pain intensities being a risk factor for poor prognosis in other musculoskeletal conditions [10].

Pain body diagrams are reliable for evaluating spatial pain extent among patients with musculoskeletal pain (e.g., shoulder, low back, but not specifically post-LLA) [11]. In other patient populations with musculoskeletal pain, a greater number of pain sites has been associated with greater healthcare utilization, sickness absenteeism, and work-related restrictions [12]. Further, a systematic review found greater extent of pain is a risk factor for poor prognosis across a range of musculoskeletal conditions [10]. To our knowledge, however, relationships between pain extent and post-amputation outcomes, such as persistent activity restrictions, remain unknown.

Following unilateral LLA, adjustment to amputation and prosthesis use are key quality-of-life determinants [13]. Greater prosthesis satisfaction and more distal LLA are associated with positive adjustment [14]. Adjustment may be better in the absence of PLP and/or RLP [8, 15], yet results are conflicting [16]. To date, however, there is little research regarding pain extent and its relationship to post-amputation adjustment and satisfaction.

This study’s primary objective was to determine if pain extent is associated with prosthetic outcomes. We hypothesized greater pain extent would be associated with greater activity restrictions, worse adjustment, and lower prosthesis satisfaction, after considering sex, age, and amputation level. Sex-related differences in pain mechanisms and outcomes are well-established [17], but under-studied post-amputation. Older age and more proximal LLA have been associated with worse prosthetic outcomes [18]. A secondary objective was to determine test-retest reliability for evaluation of pain extent post-amputation.

Methods

This online, cross-sectional study recruited participants from August 2020 to April 2021 via rehabilitation and prosthetic clinics, Amputee Support groups, online advertisements, and research databases. Participants were aged 18–75 years, English-speaking and -reading, had access to a computer and basic computer skills. Participants reported a non-congenital, unilateral LLA occurring ≥1 year prior, no acute residual limb issues (e.g., wounds), and were using a prosthesis. Adults who had participated in treatment that might affect body perception (e.g., graded motor imagery [19], mirror therapy [20]), and consequently, adjustment and satisfaction post-amputation, were excluded.

After signing informed consents, participants completed data collections using Gorilla™, a fee-for-service, web-based platform (https://gorilla.sc/) [21]. Individuals provided demographics, amputation-related, and prosthesis-related information. The Houghton Scale was administered; scores ≥9/12 suggest individuals are community-ambulators [22]. Average Socket Comfort Scores, rated 0 (most uncomfortable fit) to 10 (most comfortable fit imaginable) [23], were obtained; test-retest reliability has been reported [intraclass correlation coefficient (ICC)3,1 = 0.79, 95% confidence interval (CI): 0.67–0.86] [24].

Outcomes of interest were three psychosocial adjustment subscales (general adjustment, social adjustment, and adjustment to limitation, where higher scores are better), an activity restrictions subscale (where lower scores are better), and the functional satisfaction with prosthesis subscale (where higher scores are better), from the validated Trinity Amputation and Prosthesis Experience Scales-Revised [25]. Better TAPES-R scores have been associated with greater functional independence [28].

Number of painful regions per body diagrams (Figure 1) was summed to evaluate pain extent [26, 27]. Previous research suggests pain body diagrams with predefined regions have better test-retest reliability as compared to body diagrams using gridded transparencies (to determine the number of painful squares) [28]. Pain distributions were also explored in five distinct areas used in various ‘widespread’ pain definitions: axial pain (neck, chest, upper and lower back), right and left upper extremities, and the intact and residual lower extremities [29].

Figure 1: 
          Pain body diagrams. Participants identified all locations of pain in the past week. For widespread pain assessment, axial pain was defined as regions 3, 6, 16, 32, 35, 38, and 41. The right upper extremity regions included: 4, 7, 9, 12, 17, 19, 34, 37, 40, 43, 47, and 49, while left upper extremity regions included: 5, 8, 10, 13, 18, 20, 33, 36, 39, 42, 46, and 48. Sound and residual limbs varied based on the side amputated. Body regions 1, 2, 11, and 16 were not included when evaluating widespread pain.
Figure 1:

Pain body diagrams. Participants identified all locations of pain in the past week. For widespread pain assessment, axial pain was defined as regions 3, 6, 16, 32, 35, 38, and 41. The right upper extremity regions included: 4, 7, 9, 12, 17, 19, 34, 37, 40, 43, 47, and 49, while left upper extremity regions included: 5, 8, 10, 13, 18, 20, 33, 36, 39, 42, 46, and 48. Sound and residual limbs varied based on the side amputated. Body regions 1, 2, 11, and 16 were not included when evaluating widespread pain.

Test-retest reliability may vary based on regional involvement (e.g., better when evaluating limb pain vs. axial pain) [30] and pain intensity (e.g., better if higher) [31]. Reliability may depend on the painful sensation, e.g., ‘aching’ sensations are more reliably reported than ‘pins and needles’ [31]. Hence, a subset of participants recompleted pain body diagrams within 14 days to establish between-days, test-retest reliability for adults with LLA, given the potential for unique pain distributions and sensations, e.g., PLP, in this population. Participants and assessors were blinded to previous body diagrams.

Using SPSS 26 (IBM, Armonk, NY, USA), for participants with two days of body diagram data, test-retest reliability for number of painful sites (0–59) was evaluated using ICC3,1 (two-way mixed effects, absolute agreement, single rater) [32]. Values for the corresponding 95% CIs, standard error of measurement (SEM), and minimal detectable changes at 90% (MDC90) and 95% (MDC95) confidence levels were calculated. ICCs were interpreted as <0.50 = poor, 0.50–0.74 = fair, 0.75–0.89 = good, and ≥0.90 = excellent reliability [32]. Cohen’s Kappa was used to evaluate intra-rater agreement for reporting on pain distributions used when defining ‘widespread’ pain [33, 34]. Five linear regression models evaluated associations between pain extent and TAPES-R subscale scores, after controlling for suspected covariates (alpha ≤ 0.010).

Results

Participants

Of the 150 individuals with LLA who underwent telephone screening, 96 were eligible and interested, and 74 completed all outcome measures of interest (Figure 2). Of the 74, 54 participants had pain diagram data for two days and were included in reliability analyses. Participants were largely middle-aged adults, and of the sample, 32 participants were female (Table 1). The majority of participants had a unilateral, transtibial amputation. Trauma was the single leading cause of LLA. Participants were predominantly community-ambulators, per the Houghton Scale, and long-term prosthesis users (with a median time since amputation of 8.5 years and n = 46 reporting prosthesis use for >5 years).

Figure 2: 
            Participant inclusion flow diagram.
Figure 2:

Participant inclusion flow diagram.

Table 1:

Participant characteristics (n = 74).

Variable Median (25th, 75th percentile)
Demographics
 Sex, female* 32 (43.2%)
 Race, Caucasian* 68 (91.9%)
 Ethnicity, Non-Hispanic* 72 (97.3%)
 Age, years 58 (48, 65)
 Height, m 1.73 (1.63, 1.80)
 Weight with prosthesis, kg 86.0 (72.4, 100.9)
Amputation-related
 Type*
  Transtibial 42 (56.8%)
  Transfemoral 26 (35.1%)
  Knee disarticulation 3 (4.0%)
  Hip disarticulation 2 (2.7%)
  Rotationplasty 1 (1.4%)
 Reason*
  Trauma 27 (36.5%)
  Cancer 16 (21.6%)
  Infection 12 (16.2%)
  Dysvascular 10 (13.5%)
  Multiple or other reasons 9 (12.2%)
 Time since amputation, years 8.5 (4.0, 20.3)
Prosthesis-related
 Prosthesis experience*
  6 months–1 year 1 (1.4%)
  1–3 years 2 (2.7%)
  3–5 years 25 (33.8%)
  >5 years 46 (62.1%)
 Houghton scale, 0–12 11 (10, 12)
 Average Socket Comfort Score, 0–10 7.7 (6.7, 8.7)
  1. Participants were largely middle-aged with a unilateral transtibial or transfemoral amputation. The two most common causes of amputation were trauma and cancer, and the median time since amputation was 8.5 years. Most individuals had at least 3 years of prosthesis experience and had overall good socket comfort.

    *Data presented as n (% of sample) rather than median (25th, 75th percentile).

    Average of ‘at present’, and ‘best’ and ‘worst’ fit in the past 24 h.

    m, meters; kg, kilograms.

Outcome measures

Table 2 presents pain body diagram and TAPES-R subscale data. The median number of painful body regions was 3 (range: 0–18 sites), with n = 6, n = 8, and n = 60, reporting 0 sites, 1 site, and 2 or more painful sites, respectively. Of all body regions, phantom pain in the dorsal residual foot region was most commonly reported, affecting 28 participants. In addition to other phantom and/or residual limb pain sites (i.e., anterior residual shin, ankle, and knee, posterior residual calf), the lumbar region and anterior right shoulder were among the most commonly reported pain locations. Of the sample, 16, 4, and 4 participants had involvement of 3, 4, and 5 (out of 5) ‘widespread’ pain areas, respectively. The number of body areas affected (range: 0–5) did not differ based on level of amputation [knee disarticulation or proximal vs. transtibial or distal; X2 (5,74) = 3.21, p = 0.668]. Using the definitions of axial pain plus ≥3 quadrants with pain [29], 7 of the 74 participants had ‘widespread’ pain, while using the definition of axial pain plus pain in all four quadrants [29] suggested ‘widespread’ pain was present in four participants.

Table 2:

Outcome measures (n = 74).

Variable Median (25th, 75th percentile)
Pain body diagrams
 Total number of painful body regions, 0–59 3 (2, 7)
 Most frequently reported painful body regions*
  Dorsal residual foot 28 (37.8%)
  Anterior residual shin 21 (28.4%)
  Lumbar region 20 (27.0%)
  Anterior residual ankle 20 (27.0%)
  Anterior residual knee 19 (25.7%)
  Anterior right shoulder 14 (18.9%)
  Posterior residual calf 14 (18.9%)
Widespread pain area
 Axial 29 (39.2%)
 Right upper extremity 15 (20.3%)
 Left upper extremity 27 (36.5%)
 Sound limb 22 (29.7%)
 Residual limb
  Transtibial-level (n = 42) 25 (59.5%)
  Knee disarticulation/more proximal (n = 32) 23 (71.9%)
Total number of areas, 0–5 ¥
 0 pain areas 8 (10.8%)
  Transtibial-level 5 (11.9%)
  Knee disarticulation/more proximal 3 (9.4%)
 1 pain area 27 (36.5%)
  Transtibial-level 12 (28.6%)
  Knee disarticulation/more proximal 15 (46.9%)
 2 pain areas 15 (20.2%)
  Transtibial-level 10 (23.8%)
  Knee disarticulation/more proximal 5 (15.6%)
 3 pain areas 16 (21.6%)
  Transtibial-level 10 (23.8%)
  Knee disarticulation/more proximal 6 (18.8%)
 4 pain areas 4 (5.4%)
  Transtibial-level 3 (7.1%)
  Knee disarticulation/more proximal 1 (3.1%)
 5 pain areas 4 (5.4%)
  Transtibial-level 2 (4.8%)
  Knee disarticulation/more proximal 2 (6.3%)
TAPES-R subscale
 General adjustment, 1–4 3.8 (3.0, 4.0)
 Social adjustment, 1–4 3.8 (3.2, 4.0)
 Adjustment to limitation, 1–4 2.8 (2.4, 3.6)
 Activity restrictions, 0–2 0.7 (0.3, 1.1)
 Functional satisfaction with prosthesis, 5–15 11.0 (10.0, 14.0)
  1. Participants reported a median of three painful body regions per body diagrams, with the most frequently reported regions being the residual lower limb and lumbar spine. Total number of areas affected was not significantly different between participants with transtibial-level amputations and more proximal amputations. Adjustment, activity restrictions, and functional satisfaction with the prosthesis per the Trinity Amputation and Prosthesis Experience Scale-Revised (TAPES-R) are provided.

    *Reported as n (% of sample) rather than median (25th, 75th percentile).

    Site indicative of phantom limb pain (pain perceived as coming from the amputated portion of the limb).

    Site indicative of either phantom limb pain or residual limb pain (pain in the remaining portion of the amputated limb), dependent upon amputation level.

    ¥Age (rs = 0.10; p = 0.388) and sex [X2(5, 74) = 6.26, p = 0.282] did not significantly affect Total Number of Areas involved.

TAPES-R subscales suggest that while general and social adjustment were high (i.e., 3.8 out of 4.0), adjustment to limitation was lower at 2.8 out of 4.0. Individuals were generally a ‘little limited’ per the Activity Restrictions subscale (where 0 = not limited at all; 2 = limited a lot) and ‘satisfied’ with their prosthesis function.

Reliability

The median (25th, 75th percentile) number of days between completions of pain body diagrams was 2 (1, 8). Between-days (i.e., sessions), test-retest reliability for total number of painful body regions (0–59 sites) was good (ICC3,1 = 0.84, 95% CI: 0.73–0.90). The SEM was 1.9 regions, with MDC90 and MDC95 of 4.4 and 5.2 regions, respectively. Between-days, intra-rater agreement for widespread pain areas was 63.4, 75.4, 71.2, 50.4, and 66.7%, for axial, right upper extremity, left upper extremity, intact lower-extremity, and residual lower-extremity, respectively. The between-days, intra-rater agreements were not significantly different between the widespread pain areas, as 95% confidence intervals [calculated around the difference between two Cohen’s kappa statistics (e.g., axial vs. right upper extremity)], included zero, indicating no difference [35].

Regression analyses

Table 3 provides the five linear regression models. All but one model (i.e., Social Adjustment) met parametric assumptions; residuals from the Social Adjustment model violated the assumptions of normality even after outlier removal, but robust standard error estimates confirmed model findings. Covariates were independently associated with the TAPES-R Activity Restriction subscale (adjusted R2 = 0.271, p < 0.001); however, associations between covariates and all other subscales did not reach significance (p > 0.010). Beyond covariates, the total number of painful body regions (pain extent) was significantly associated with all TAPES-R subscales (p < 0.001–0.006), with the exception of Social Adjustment (p = 0.764). Specifically, the total number of painful body regions independently explained 14.5% of the variance in Functional Satisfaction with the Prosthesis, 11.8% of the variance in Adjustment to Limitation, 11.6% of the variance in General Adjustment, and 7.4% of the variance in Activity Restriction. Each additional painful body region was associated with about a 0.26-point lower Functional Satisfaction with Prosthesis score, a 0.04-point lower General Adjustment score, a 0.06-point lower Adjustment to Limitation score, and a 0.03-point greater Activity Restriction score.

Table 3:

Associations between pain site involvement and prosthetic outcomes (n = 74).

Model Block statistics Individual predictor statistics
R2 adjR2 ΔR2 p β SE p
TAPES-R general adjustment
Block 1 0.074 0.034 0.146
Block 2 0.189 0.142 0.116 0.002*
 Age −0.001 0.004 0.750
 Sex −0.039 0.110 0.726
 Amputation level −0.224 0.109 0.043
 Number of painful body regions (0–59) −0.039 0.012 0.002*
TAPES-R social adjustment
Block 1 0.054 0.013 0.271
Block 2 0.055 <0.001 0.001 0.764
 Age 0.002 0.005 0.752
 Sex −0.234 0.127 0.070
 Amputation level 0.018 0.125 0.884
 Number of painful body regions (0–59) −0.004 0.014 0.764
TAPES-R adjustment to limitation
Block 1 0.137 0.100 0.015
Block 2 0.255 0.212 0.118 0.002*
 Age −0.010 0.007 0.145
 Sex −0.202 0.168 0.232
 Amputation level −0.398 0.166 0.019
 Number of painful body regions (0–59) −0.062 0.019 0.002*
TAPES-R activity restriction
Block 1 0.300 0.270 <0.001*
Block 2 0.374 0.338 0.074 0.006*
 Age 0.009 0.004 0.021
 Sex 0.232 0.091 0.013
 Amputation level 0.363 0.090 <0.001*
 Number of painful body regions (0–59) 0.029 0.010 0.006*
TAPES-R functional satisfaction with prosthesis
Block 1 0.115 0.077 0.036
Block 2 0.260 0.217 0.145 <0.001*
 Age −0.025 0.025 0.313
 Sex 0.179 0.623 0.774
 Amputation level −1.483 0.614 0.018
 Number of painful body regions (0–59) −0.256 0.070 <0.001*
  1. Regression modeling results are provided, where age, sex (0 = female, 1 = male), and amputation level (0 = transtibial, 1 = knee disarticulation or more proximal) were entered in Block 1. Block 2 included the aforementioned covariates plus the total number of painful body regions (independent variables) to determine the total amount of variance explained by the extent of pain, above and beyond covariates. Dependent variables were TAPES-R subscale scores.

    adj, adjusted; ΔR2, R2 change; β, unstandardized beta coefficient; TAPES-R, Trinity Amputation and Prosthesis Experience Scale-Revised; SE, standard error.

    *p ≤ 0.010.

Discussion

Our findings align with prior work suggesting pain is a significant issue for adults with LLA [7], as 81.1% of our participants reported multisite pain and only 8.1% were pain-free. To our knowledge, this is the first study to evaluate widespread pain among adults with LLA and depending on the definition used [29], we estimate that somewhere between 5.4 and 9.5% of adults with LLA may have widespread pain (which is similar to prevalence reported among individuals with fibromyalgia syndrome, i.e., 4.9–8.3%) [29]. A greater spatial extent of pain, i.e., more painful regions (range 0–59), was associated with greater activity restrictions, as well as worse general adjustment, adjustment to limitations, and prosthesis satisfaction, after considering age, sex, and amputation level. In fact, the extent of pain explained more of the variance in prosthetic outcomes than non-modifiable covariates (i.e., sex, age, and amputation level), suggesting reducing and/or preventing the extent of pain may be an under-targeted pathway for enhancing post-amputation outcomes. Our cross-sectional findings of associations between pain extent and post-amputation outcomes warrant future longitudinal research to determine if pain extent is a strong predictor of long-term outcomes, including post-amputation adjustment, satisfaction, activity restrictions, and participation. Ultimately, reducing the extent of pain post-amputation may be a pathway for improving mediocre post-amputation outcomes [36], [37], [38].

Among middle-aged adults with non-congenital, unilateral LLA, we found greater pain extent to be associated with worse prosthesis functional satisfaction, general adjustment, and adjustment to limitations, as well as greater activity restrictions (per the TAPES-R). Our findings are particularly relevant given mixed research results regarding relationships between pain presence and post-amputation activity restrictions, adjustment, and prosthesis satisfaction [8, 15, 16, 39]. For example, Desmond and colleagues reported middle- and older-aged adults with unilateral LLA (n = 72) experiencing phantom and/or residual limb pain (in the week prior) had lower prosthesis satisfaction and adjustment to limitation, but similar activity restrictions and general and social adjustment per the TAPES-R, as compared to pain-free peers (n = 17) [15]. Gallagher et al. reported middle-aged adults with phantom or RLP (n = 104) had lower adjustment to limitations but similar general and social adjustment and functional prosthesis satisfaction as compared to peers without amputated-site pain [8]. Sinha and colleagues reported that neither phantom nor RLP presence were important factors in post-amputation adjustment nor prosthesis satisfaction using an exploratory stepwise analysis approach among adults with non-congenital, unilateral LLA (n = 368) [16]. Thus, perhaps extent of pain as obtained with body diagrams (which is a continuous variable), is a more sensitive measure than presence or absence of pain, and consequently, may help to differentiate among groups with varying post-amputation outcomes.

Given our sample demographics, findings are not generalizable to adults with congenital or bilateral LLA nor those with upper limb amputations. A few covariates were considered in our analyses (i.e., sex, age, and amputation level), but given the preliminary nature of this study, we are underpowered to definitively determine if covariates are contributors to post-amputation activity restrictions, adjustment, and prosthesis satisfaction. Other covariates (e.g., amputation etiology; surgical history; body image disturbance; prosthesis and assistive device use; comorbidities; use of prosthetic test sockets) [16, 40], [41], [42], [43] that may impact post-amputation outcomes should be considered in future, large-scale studies; such studies may comprehensively evaluate pain, including PLP and RLP intensity, frequency, and bothersomeness, to determine the strongest prognostic factors for poor outcomes. Longitudinal study designs are encouraged to allow assessment of stability of modifiable post-amputation factors (e.g., pain extent, body image disturbance, prosthesis use), as well as evaluation of causal relationships between such factors and post-amputation outcomes.

While we found ‘good’ reliability for evaluating the spatial extent of pain via body diagrams, reliability may have been negatively affected by (a) daily pain fluctuations (e.g., low back pain only in the evening after prosthesis use rather than constantly present) as time-of-day for administration was not controlled between sessions, and (b) overall mild [44] pain intensities reported in our sample of convenience [31]. Further, the sampling strategy may explain mild activity restrictions, greater adjustment and prosthesis satisfaction, in our sample. Future research among larger samples of care-seeking adults post-amputation may enable more granular evaluations of intra-individual, between-days agreement in reporting pain in various regions (e.g., phantom limb vs. residual limb vs. low back) [30], as well as determination of associations between pain and activity restrictions, adjustment, and satisfaction. But as a start, evaluation of test-retest reliability of widespread pain areas suggests moderate-to-substantial intra-rater agreement for axial, right upper extremity, left upper extremity, intact lower-extremity, and residual lower-extremity pain areas, and similar intra-rater agreement among these widespread pain areas among individuals post-LLA. Of note, defined areas for ‘widespread pain’ are not universal and may exclude craniofacial, abdominal, and genitourinary regions, and known discrepancies regarding what constitutes buttock (i.e., lower extremity) vs. low back pain (i.e., axial) are acknowledged [29].

In summary, among adults with non-congenital, unilateral LLA using a prosthesis, greater pain extent is associated with greater activity restriction, worse post-amputation adjustment, and lower prosthesis satisfaction, after considering age, sex, and amputation level. Pain body diagrams have good test-retest reliability for assessing pain extent post-amputation using 59 predefined regions and appear to have moderate-to-substantial intra-rater agreement for evaluating widespread pain. Thus, pain body diagrams are encouraged in clinical practice and future pain-related research post-amputation, especially since 80% of participants with unilateral LLA in this study reported multisite pain, and up to 9.5% reported widespread pain, of which, neither may be captured by simply asking about amputated-site (e.g., residual, phantom limb) pain. Pending future studies establishing causal relationships between pain extent and post-amputation outcomes (e.g., activity restrictions, adjustment, and prosthesis satisfaction), interventions targeting post-amputation pain prevention and alleviation may be an untapped pathway for improving post-amputation outcomes.


Corresponding author: Jaclyn Megan Sions PT, DPT, PhD, Department of Physical Therapy, University of Delaware, 540 South College Avenue, Suite 210JJ, Newark, DE19713, USA, Phone: +1 302 831 7231, E-mail:

An abstract related to this work has been submitted and accepted for presentation to the American Physical Therapy Association’s Combined Sections Meeting to be held in San Antonio, Texas, USA from 02-02-2022 to 02-05-2022.


  1. Research funding: This work was supported by the Eunice Kennedy Shriver National Institute of Child Health and Human Development of the National Institutes of Health [T32HD007490], the Foundation for Physical Therapy Research Promotion of Doctoral Studies I and II Scholarships awarded to Dr. Beisheim-Ryan, and the Independence Prosthetics-Orthotics, Inc. Postdoctoral Researcher Fund. The content is solely the responsibility of the authors and does not necessarily represent the official views of the funding institutions.

  2. Author contributions: All authors have accepted responsibility for the entire content of this manuscript and approved its submission.

  3. Competing interests: Authors state no conflict of interest.

  4. Informed consent: Informed consent has been obtained from all individuals included in this study.

  5. Ethical approval: Research involving human subjects complied with all relevant national regulations, institutional policies and is in accordance with the tenets of the Helsinki Declaration (as amended in 2013), and has been approved by the authors’ Institutional Review Board (IRB #1611862).

References

1. Thapa, S, Shmerling, RH, Bean, JF, Cai, Y, Leveille, SG. Chronic multisite pain: evaluation of a new geriatric syndrome. Aging Clin Exp Res 2019;31:1129–37. https://doi.org/10.1007/s40520-018-1061-3.Search in Google Scholar PubMed PubMed Central

2. Finney, A, Dziedzic, KS, Lewis, M, Healey, E. Multisite peripheral joint pain: a cross-sectional study of prevalence and impact on general health, quality of life, pain intensity and consultation behaviour. BMC Musculoskelet Disord 2017;18:535. https://doi.org/10.1186/s12891-017-1896-3.Search in Google Scholar PubMed PubMed Central

3. Beisheim, EH, Seth, M, Horne, JR, Hicks, GE, Pohlig, RT, Sions, JM. Sex-specific differences in multisite pain presentation among adults with lower-limb loss. Pain Pract 2021;21:419–27. https://doi.org/10.1111/papr.12969.Search in Google Scholar PubMed PubMed Central

4. Gailey, R, Allen, K, Castles, J, Kucharik, J, Roeder, M. Review of secondary physical conditions associated with lower-limb amputation and long-term prosthesis use. J Rehabil Res Dev 2008;45:15–29. https://doi.org/10.1682/jrrd.2006.11.0147.Search in Google Scholar PubMed

5. Doring, K, Trost, C, Hofer, C, Salzer, M, Kelaridis, T, Windhager, R, et al.. How common are chronic residual limb pain, phantom pain, and back pain more than 20 years after lower limb amputation for malignant tumors? Clin Orthop Relat Res 2021;479:2036–44. https://doi.org/10.1097/CORR.0000000000001725.Search in Google Scholar PubMed PubMed Central

6. Devan, H, Tumilty, S, Smith, C. Physical activity and lower-back pain in persons with traumatic transfemoral amputation: a national cross-sectional survey. J Rehabil Res Dev 2012;49:1457–66. https://doi.org/10.1682/jrrd.2011.09.0155.Search in Google Scholar PubMed

7. Radhakrishnan, S, Kohler, F, Gutenbrunner, C, Jayaraman, A, Li, J, Pieber, K, et al.. The use of the International Classification of Functioning, Disability and Health to classify the factors influencing mobility reported by persons with an amputation: an international study. Prosthet Orthot Int 2017;41:412–9. https://doi.org/10.1177/0309364616652016.Search in Google Scholar PubMed

8. Gallagher, P, Allen, D, Maclachlan, M. Phantom limb pain and residual limb pain following lower limb amputation: a descriptive analysis. Disabil Rehabil 2001;23:522–30. https://doi.org/10.1080/09638280010029859.Search in Google Scholar PubMed

9. Damiani, C, Pournajaf, S, Goffredo, M, Proietti, S, Denza, G, Rosa, B, et al.. Community ambulation in people with lower limb amputation: an observational cohort study. Medicine (Baltim) 2021;100:e24364. https://doi.org/10.1097/MD.0000000000024364.Search in Google Scholar PubMed PubMed Central

10. Artus, M, Campbell, P, Mallen, CD, Dunn, KM, van der Windt, DAW. Generic prognostic factors for musculoskeletal pain in primary care: a systematic review. BMJ Open 2017;7:e012901. https://doi.org/10.1136/bmjopen-2016-012901.Search in Google Scholar PubMed PubMed Central

11. Southerst, D, Cote, P, Stupar, M, Stern, P, Mior, S. The reliability of body pain diagrams in the quantitative measurement of pain distribution and location in patients with musculoskeletal pain: a systematic review. J Manip Physiol Ther 2013;36:450–9. https://doi.org/10.1016/j.jmpt.2013.05.021.Search in Google Scholar PubMed

12. Fernandes, RCP, Burdorf, A. Associations of multisite pain with healthcare utilization, sickness absence and restrictions at work. Int Arch Occup Environ Health 2016;89:1039–46. https://doi.org/10.1007/s00420-016-1141-7.Search in Google Scholar PubMed PubMed Central

13. Sinha, R, van den Heuvel, WJ, Arokiasamy, P, van Dijk, JP. Influence of adjustments to amputation and artificial limb on quality of life in patients following lower limb amputation. Int J Rehabil Res 2014;37:74–9. https://doi.org/10.1097/MRR.0000000000000038.Search in Google Scholar PubMed

14. Horgan, O, MacLachlan, M. Psychosocial adjustment to lower-limb amputation: a review. Disabil Rehabil 2004;26:837–50. https://doi.org/10.1080/09638280410001708869.Search in Google Scholar PubMed

15. Desmond, D, Gallagher, P, Henderson-Slater, D, Chatfield, R. Pain and psychosocial adjustment to lower limb amputation amongst prosthesis users. Prosthet Orthot Int 2008;32:244–52. https://doi.org/10.1080/03093640802067046.Search in Google Scholar PubMed

16. Sinha, R, van den Heuvel, WJ, Arokiasamy, P. Adjustments to amputation and an artificial limb in lower limb amputees. Prosthet Orthot Int 2014;38:115–21. https://doi.org/10.1177/0309364613489332.Search in Google Scholar PubMed

17. Aternali, A, Katz, J. Recent advances in understanding and managing phantom limb pain. F1000Res 2019;8:F1000 Faculty Rev-1167. https://doi.org/10.12688/f1000research.19355.1.Search in Google Scholar PubMed PubMed Central

18. Karaali, E, Duramaz, A, Ciloglu, O, Yalin, M, Atay, M, Aslantas, FC. Factors affecting activities of daily living, physical balance, and prosthesis adjustment in non-traumatic lower limb amputees. Turk J Phys Med Rehabil 2020;66:405–12. https://doi.org/10.5606/tftrd.2020.4623.Search in Google Scholar PubMed PubMed Central

19. Bowering, KJ, O’Connell, NE, Tabor, A, Catley, MJ, Leake, HB, Moseley, GL, et al.. The effects of graded motor imagery and its components on chronic pain: a systematic review and meta-analysis. J Pain 2013;14:3–13. https://doi.org/10.1016/j.jpain.2012.09.007.Search in Google Scholar PubMed

20. Foell, J, Bekrater-Bodmann, R, Diers, M, Flor, H. Mirror therapy for phantom limb pain: brain changes and the role of body representation. Eur J Pain 2014;18:729–39. https://doi.org/10.1002/j.1532-2149.2013.00433.x.Search in Google Scholar PubMed

21. Anwyl-Irvine, AL, Massonnie, J, Flitton, A, Kirkham, N, Evershed, JK. Gorilla in our midst: an online behavioral experiment builder. Behav Res Methods 2020;52:388–407. https://doi.org/10.3758/s13428-019-01237-x.Search in Google Scholar PubMed PubMed Central

22. Wong, CK, Gibbs, W, Chen, ES. Use of the Houghton Scale to classify community and household walking ability in people with lower-limb amputation: criterion-related validity. Arch Phys Med Rehabil 2016;97:1130–6. https://doi.org/10.1016/j.apmr.2016.01.022.Search in Google Scholar PubMed

23. Hanspal, RS, Fisher, K, Nieveen, R. Prosthetic socket fit comfort score. Disabil Rehabil 2003;25:1278–80. https://doi.org/10.1080/09638280310001603983.Search in Google Scholar PubMed

24. Hafner, BJ, Morgan, SJ, Askew, RL, Salem, R. Psychometric evaluation of self-report outcome measures for prosthetic applications. J Rehabil Res Dev 2016;53:797–812. https://doi.org/10.1682/JRRD.2015.12.0228.Search in Google Scholar PubMed PubMed Central

25. Gallagher, P, Franchignoni, F, Giordano, A, MacLachlan, M. Trinity amputation and prosthesis experience scales: a psychometric assessment using classical test theory and rasch analysis. Am J Phys Med Rehabil 2010;89:487–96. https://doi.org/10.1097/PHM.0b013e3181dd8cf1.Search in Google Scholar PubMed

26. Margolis, RB, Tait, RC, Krause, SJ. A rating system for use with patient pain drawings. Pain 1986;24:57–65. https://doi.org/10.1016/0304-3959(86)90026-6.Search in Google Scholar PubMed

27. Margolis, RB, Chibnall, JT, Tait, RC. Test-retest reliability of the pain drawing instrument. Pain 1988;33:49–51. https://doi.org/10.1016/0304-3959(88)90202-3.Search in Google Scholar PubMed

28. Roach, KE, Brown, MD, Dunigan, KM, Kusek, CL, Walas, M. Test-retest reliability of patient reports of low back pain. J Orthop Sports Phys Ther 1997;26:253–9. https://doi.org/10.2519/jospt.1997.26.5.253.Search in Google Scholar PubMed

29. Butler, S, Landmark, T, Glette, M, Borchgrevink, P, Woodhouse, A. Chronic widespread pain-the need for a standard definition. Pain 2016;157:541–3. https://doi.org/10.1097/j.pain.0000000000000417.Search in Google Scholar PubMed

30. Ohnmeiss, DD. Repeatability of pain drawings in a low back pain population. Spine (Phila Pa 1976) 2000;25:980–8. https://doi.org/10.1097/00007632-200004150-00014.Search in Google Scholar PubMed

31. Love, A, Leboeuf, C, Crisp, TC. Chiropractic chronic low back pain sufferers and self-report assessment methods. Part I. A reliability study of the Visual Analogue Scale, the Pain Drawing and the McGill Pain Questionnaire. J Manip Physiol Ther 1989;12:21–5.Search in Google Scholar

32. Koo, TK, Li, MY. A guideline of selecting and reporting intraclass correlation coefficients for reliability research. J Chiropr Med 2016;15:155–63. https://doi.org/10.1016/j.jcm.2016.02.012.Search in Google Scholar PubMed PubMed Central

33. Watson, PF, Petrie, A. Method agreement analysis: a review of correct methodology. Theriogenology 2010;73:1167–79. https://doi.org/10.1016/j.theriogenology.2010.01.003.Search in Google Scholar PubMed

34. Landis, JR, Koch, GG. The measurement of observer agreement for categorical data. Biometrics 1977;33:159–74.10.2307/2529310Search in Google Scholar

35. Tan, SH, Tan, SB. The correct interpretation of confidence intervals. Proc Singap Healthc 2010;19:276–8. https://doi.org/10.1177/201010581001900316.Search in Google Scholar

36. Hofstad, CJ, Bongers, KTJ, Didden, M, van Ee, RF, Keijsers, NLW. Maximal walking distance in persons with a lower limb amputation. Sensors (Basel) 2020;20:6770. https://doi.org/10.3390/s20236770.Search in Google Scholar PubMed PubMed Central

37. Davie-Smith, F, Paul, L, Nicholls, N, Stuart, WP, Kennon, B. The impact of gender, level of amputation and diabetes on prosthetic fit rates following major lower extremity amputation. Prosthet Orthot Int 2017;41:19–25. https://doi.org/10.1177/0309364616628341.Search in Google Scholar PubMed PubMed Central

38. Darter, BJ, Hawley, CE, Armstrong, AJ, Avellone, L, Wehman, P. Factors influencing functional outcomes and return-to-work after amputation: a review of the literature. J Occup Rehabil 2018;28:656–65. https://doi.org/10.1007/s10926-018-9757-y.Search in Google Scholar PubMed PubMed Central

39. Durmus, D, Safaz, I, Adiguzel, E, Uran, A, Sarisoy, G, Goktepe, AS, et al.. The relationship between prosthesis use, phantom pain and psychiatric symptoms in male traumatic limb amputees. Compr Psychiatry 2015;59:45–53. https://doi.org/10.1016/j.comppsych.2014.10.018.Search in Google Scholar PubMed

40. Murray, CD, Fox, J. Body image and prosthesis satisfaction in the lower limb amputee. Disabil Rehabil 2002;24:925–31. https://doi.org/10.1080/09638280210150014.Search in Google Scholar PubMed

41. Batten, H, Lamont, R, Kuys, S, McPhail, S, Mandrusiak, A. What are the barriers and enablers that people with a lower limb amputation experience when walking in the community? Disabil Rehabil 2020;42:3481–7. https://doi.org/10.1080/09638288.2019.1597177.Search in Google Scholar PubMed

42. Luza, LP, Ferreira, EG, Minsky, RC, Pires, GKW, da Silva, R. Psychosocial and physical adjustments and prosthesis satisfaction in amputees: a systematic review of observational studies. Disabil Rehabil Assist Technol 2020;15:582–9. https://doi.org/10.1080/17483107.2019.1602853.Search in Google Scholar PubMed

43. Aydin, A, Caglar Okur, S. Effects of test socket on pain, prosthesis satisfaction, and functionality in patients with transfemoral and transtibial amputations. Med Sci Monit 2018;24:4031–7. https://doi.org/10.12659/MSM.910858.Search in Google Scholar PubMed PubMed Central

44. Treede, RD, Rief, W, Barke, A, Aziz, Q, Bennett, MI, Benoliel, R, et al.. Chronic pain as a symptom or a disease: the IASP Classification of Chronic Pain for the International Classification of Diseases (ICD-11). Pain 2019;160:19–27. https://doi.org/10.1097/j.pain.0000000000001384.Search in Google Scholar PubMed

Received: 2021-07-22
Accepted: 2022-01-11
Published Online: 2022-02-01
Published in Print: 2022-07-26

© 2022 Walter de Gruyter GmbH, Berlin/Boston

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