Evaluation of Measures of Upper Limb Functioning and Disability in People With Parkinson Disease:...

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REVIEW ARTICLE Evaluation of Measures of Upper Limb Functioning and Disability in People With Parkinson Disease: A Systematic Review Elizabeth L. Proud, MPhysio (Neuro), a Kimberly J. Miller, PhD, a,b Belinda Bilney, PhD, a Sulakshana Balachandran, BPhty(Hon1), a Jennifer L. McGinley, PhD, a Meg E. Morris, PhD c From the a Department of Physiotherapy, School of Health Sciences, The University of Melbourne, Victoria, Australia; b Department of Physical Therapy, University of British Columbia, Vancouver, British Columbia, Canada; and c Department of Physiotherapy, School of Allied Health, La Trobe University, Victoria, Australia. Abstract Objectives: To identify measurement tools used for upper limb evaluation in people with Parkinson disease (PD), to summarize the content of each tool using the International Classification of Functioning, Disability and Health, and to examine the reliability, validity, clinical utility, and responsiveness of the measurement tools specific to this clinical group. Data Sources: Two systematic searches of online databases included articles published from inception to November 2013. Study Selection: Search 1 identified upper limb measures. Search 2 retrieved studies investigating the measurement properties of these tools in people with PD. Data Extraction: Independent reviewers screened articles and extracted data, classified measurement tool content on the basis of the International Classification of Functioning, Disability and Health content domains, and applied both the COnsensus-based Standards for the selection of health status Measurement INstruments checklist to evaluate the study’s methodological quality and a second checklist by Terwee et al to assess the measurement tool’s quality. A third reviewer adjudicated differences between reviewers. Information on clinical utility was also compiled. Data Synthesis: The 18 identified measures included PD-specific scales, generic measures, and tools developed for other clinical populations; most measures evaluated impairments and/or activity limitations. Measurement properties of 10 of the 18 identified measures were evaluated in people with PD. No high-quality studies investigated validity or responsiveness. High-quality evidence supported the interrater reliability of some Unified Parkinson’s Disease Rating Scale subtests, and lower quality studies provided limited evidence for the test-retest reliability of measures evaluating fine hand function and bradykinesia. Conclusions: There are relatively few high-quality studies to support the measurement properties, particularly the validity and responsiveness, of tools currently used to evaluate upper limb disability and function in people with PD. Further research is needed to inform measurement tool selection and treatment evaluation in this clinical group. Archives of Physical Medicine and Rehabilitation 2014;-:------- ª 2014 by the American Congress of Rehabilitation Medicine Upper limb movement disorders may be present in people with Parkinson disease (PD) from diagnosis, with impairments such as micrographia or resting tremor often being the first sign of the disease. 1 Difficulties in manipulating objects with appropriate speed and dexterity are also reported. 2-4 As the disease progresses, these upper limb disorders can lead to limitations in work, rec- reation, 5 and daily tasks such as dressing and eating. 6 An important component of evidence-based practice is the use of measurement tools with strong measurement properties to quantify disease progression and treatment outcomes 7 ; however, current guidelines for physiotherapy and occupational therapy management of people with PD contain few recommendations to assist in the selection of suitable upper limb measurement tools. 6,8,9 Physiotherapists and occupational therapists who Preliminary results presented as a poster to the World Parkinson Congress, October 1-4, 2013, Montreal, Quebec, Canada. Disclosures: none. 0003-9993/14/$36 - see front matter ª 2014 by the American Congress of Rehabilitation Medicine http://dx.doi.org/10.1016/j.apmr.2014.09.016 Archives of Physical Medicine and Rehabilitation journal homepage: www.archives-pmr.org Archives of Physical Medicine and Rehabilitation 2014;-:-------

Transcript of Evaluation of Measures of Upper Limb Functioning and Disability in People With Parkinson Disease:...

edicine and Rehabilitation

Archives of Physical M journal homepage: www.archives-pmr.org

Archives of Physical Medicine and Rehabilitation 2014;-:-------

REVIEW ARTICLE

Evaluation of Measures of Upper Limb Functioningand Disability in People With Parkinson Disease:A Systematic Review

Elizabeth L. Proud, MPhysio (Neuro),a Kimberly J. Miller, PhD,a,b Belinda Bilney, PhD,a

Sulakshana Balachandran, BPhty(Hon1),a Jennifer L. McGinley, PhD,a Meg E. Morris, PhDc

From the aDepartment of Physiotherapy, School of Health Sciences, The University of Melbourne, Victoria, Australia; bDepartment of PhysicalTherapy, University of British Columbia, Vancouver, British Columbia, Canada; and cDepartment of Physiotherapy, School of Allied Health, LaTrobe University, Victoria, Australia.

Abstract

Objectives: To identify measurement tools used for upper limb evaluation in people with Parkinson disease (PD), to summarize the content of

each tool using the International Classification of Functioning, Disability and Health, and to examine the reliability, validity, clinical utility, and

responsiveness of the measurement tools specific to this clinical group.

Data Sources: Two systematic searches of online databases included articles published from inception to November 2013.

Study Selection: Search 1 identified upper limb measures. Search 2 retrieved studies investigating the measurement properties of these tools in

people with PD.

Data Extraction: Independent reviewers screened articles and extracted data, classified measurement tool content on the basis of the International

Classification of Functioning, Disability and Health content domains, and applied both the COnsensus-based Standards for the selection of health

status Measurement INstruments checklist to evaluate the study’s methodological quality and a second checklist by Terwee et al to assess the

measurement tool’s quality. A third reviewer adjudicated differences between reviewers. Information on clinical utility was also compiled.

Data Synthesis: The 18 identified measures included PD-specific scales, generic measures, and tools developed for other clinical populations;

most measures evaluated impairments and/or activity limitations. Measurement properties of 10 of the 18 identified measures were evaluated in

people with PD. No high-quality studies investigated validity or responsiveness. High-quality evidence supported the interrater reliability of some

Unified Parkinson’s Disease Rating Scale subtests, and lower quality studies provided limited evidence for the test-retest reliability of measures

evaluating fine hand function and bradykinesia.

Conclusions: There are relatively few high-quality studies to support the measurement properties, particularly the validity and responsiveness, of

tools currently used to evaluate upper limb disability and function in people with PD. Further research is needed to inform measurement tool

selection and treatment evaluation in this clinical group.

Archives of Physical Medicine and Rehabilitation 2014;-:-------

ª 2014 by the American Congress of Rehabilitation Medicine

Upper limb movement disorders may be present in people withParkinson disease (PD) from diagnosis, with impairments such asmicrographia or resting tremor often being the first sign of thedisease.1 Difficulties in manipulating objects with appropriatespeed and dexterity are also reported.2-4 As the disease progresses,

Preliminary results presented as a poster to the World Parkinson Congress, October 1-4, 2013,

Montreal, Quebec, Canada.

Disclosures: none.

0003-9993/14/$36 - see front matter ª 2014 by the American Congress of Re

http://dx.doi.org/10.1016/j.apmr.2014.09.016

these upper limb disorders can lead to limitations in work, rec-reation,5 and daily tasks such as dressing and eating.6

An important component of evidence-based practice is the useof measurement tools with strong measurement properties toquantify disease progression and treatment outcomes7; however,current guidelines for physiotherapy and occupational therapymanagement of people with PD contain few recommendations toassist in the selection of suitable upper limb measurementtools.6,8,9 Physiotherapists and occupational therapists who

habilitation Medicine

2 E.L. Proud et al

completed a recent survey of current practice reported anonuniform approach to clinical upper limb measurement inpeople with PD.10 Respondents used a range of measures in theirpractice including generic manual dexterity tests and toolsdeveloped for other clinical populations, and some survey re-spondents were unable to identify upper limb measures suitableto their needs.10

When selecting measurement tools, it is necessary to have anunderstanding of their measurement properties, namely, theirreliability, validity, and responsiveness.11 Knowledge of clinicalutility can also guide clinicians in choosing measurement toolsbest suited to their clinical situation. Because measurement toolsare administered to inform treatment decisions, it is important toidentify the impairments, activity limitations, and participationrestrictions that can be quantified by individual measures. TheInternational Classification of Functioning, Disability and Health(ICF)12 provides a framework for the identification and compari-son of measurement tool content.13

This review provides information for clinicians and researchersto inform the selection of appropriate tools for upper limb mea-surement in people with PD. The specific aims of this review wereto (1) identify measures used to evaluate upper limb impairments,activity limitations, or participation restrictions in people with PD;(2) investigate the measurement properties and clinical utility ofthese measures in this population; and (3) examine and comparethe content of the reported measures using the ICF framework.

Methods

The review was conducted in 2 parts: an initial search to identifyupper limb measures described in the PD literature and a secondsearch to retrieve studies investigating the measurement propertiesof these tools in this clinical group. Both searches were conductedin accordance with the Preferred Reporting Items for Systematicreviews and Meta-Analyses protocol.14

Part 1: Identification of upper limb measurementtools

Search strategyA search of 9 electronic databases (ISI Web of Science, MED-LINE ISI, EMBASE, Scopus, PubMed, CINAHL, PsycINFO,PEDro, and OT Seeker) was conducted in July 2012 and updatedin November 2013. The search strategy combined 3 sets of terms(1) parkinson OR “parkinson* disease”; (2) “upper limb” OR“upper extremity” OR arm OR hand; and (3) measure* ORassess* OR outcome OR tool OR instrument* OR dexterity ORdisability OR ADL and was individualized for each database.Medical Subject Headings were used when available, and nolimits were placed on publication date. The search strategy forScopus is provided in supplemental appendix S1 (available online

List of abbreviations:

CAPIT Core Assessment Program for Intracerebral

Transplantations

COSMIN COnsensus-based Standards for the selection of health

status Measurement INstruments

DASH Disabilities of the Arm, Shoulder and Hand

ICF International Classification of Functioning, Disability

and Health

PD Parkinson disease

UPDRS Unified Parkinson’s Disease Rating Scale

only at http://www.archives-pmr.org/). Reviews retrieved from theCochrane Database of Systematic Reviews using the search term“parkinson disease” were hand searched for any further studiesreporting upper limb outcomes.

EligibilityStudies were included if they (1) were published in English; (2)included participants diagnosed with idiopathic PD; (3) evaluatedthe effects of treatment or disease progression; (4) quantifiedupper limb impairments, activity limitations, and/or participationrestrictions; and (5) described a measurement method that couldbe replicated in clinical practice. Studies were excluded if thearticle text or references provided insufficient detail of the mea-surement tool or the test protocol to allow the test to be repeated.Studies using laboratory-based measures such as kinematic anal-ysis, which were considered to be beyond the scope of clinicalpractice, or obsolete commercially produced tools were alsoexcluded. Letters, editorials, literature reviews, and conferenceabstracts were excluded.

Screening and data extractionOnce duplicates were removed, titles and abstracts were screenedby one reviewer (E.P.). Two reviewers (S.B., E.P.) completed full-text screening and data extraction from included studies, with athird reviewer (K.M.) available to resolve differences.

Data extracted included participant characteristics, disease-related data, type of intervention, and details of the measurementtools used.

ICF LinkingThe content of each measure was independently examined byK.M. and E.P. using the ICF framework,12 with B.B. available toadjudicate discrepancies. Using established linking rules, mean-ingful concepts contained in individual test items were identifiedand linked to the relevant ICF classification code(s).13,15 To pro-vide a more detailed description, tool concepts were linked to thefourth-level subcategory when possible.

Clinical utilityClinical utility includes considerations relating to equipmentneeds and cost, administration time requirements, and necessityfor training.16,17 Data pertaining to the clinical utility of eachmeasure were collated by E.P. from journal articles, test manuals,and communication with equipment suppliers.

Part 2: Measurement properties of upper limb tools

Search strategyA search for studies investigating 1 or more measurement prop-erties of the tools identified in part 1 was completed in February2013 and updated in November 2013. Five electronic databaseswere searched (ISI Web of Science, MEDLINE ISI, Scopus,CINAHL, and PsycINFO). The search combined (1) the termsdyskinesia* OR hyperkinesia* OR hypokinesia OR tremor OR“activities of daily living” OR “muscle rigidity” OR movementOR “motor performance” OR “motor activity” OR dexterity ORhandwriting; (2) parkinson* disease; (3) the full name of each tooland any commonly used abbreviations, for example, “UnifiedParkinson’s Disease Rating Scale” OR UPDRS, and also “graphicstablet” OR “digitizing tablet”; and (4) a search filter originallydesigned to identify studies investigating the measurement prop-erties of health-related patient-reported outcomes listed in

www.archives-pmr.org

Fig 1 Flowchart showing selection process for searches 1 and 2.

Upper limb measures in Parkinson disease 3

PubMed.18 This filter has also been applied to other electronicdatabases in reviews of performance-based measures.19 MedicalSubject Headings were applied when available. All years wereincluded for each database, and studies were limited to those inEnglish. The search strategy for Scopus is provided insupplemental data (available online only at http://www.archives-pmr.org/). The reference lists of included articles were alsosearched for further relevant studies.

EligibilityStudies were included if (1) the participants were diagnosed withidiopathic PD; and (2) the purpose of the study was to evaluate themeasurement properties of measurement tools identified in part 1.Conference abstracts, review articles, letters, and editorialswere excluded.

Screening and data extraction

After duplicates were removed, 2 reviewers (E.P. and B.B.)independently screened titles and abstracts, and included full-textarticles, with K.M. available for adjudication. Data extractedindependently from eligible studies by the same reviewersincluded age and disease characteristics of participants, details ofmeasurement properties evaluated, and study results.

Quality assessmentTwo methods of quality assessment were completed indepen-dently by E.P. and B.B., with K.M. available to resolve differ-ences. A checklist developed by Terwee et al20 was used toapply quality criteria to the study results for investigated mea-surement properties (reliability, validity, and responsiveness)specific to each measurement tool, whereas the methodologicalquality of each study was evaluated using the COnsensus-basedStandards for the selection of health status MeasurementINstruments (COSMIN) checklist.21 Scoring for the COSMIN

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checklist used a 4-point system (ranging from “excellent” to“poor”) to rate the methodological quality of the study againstdefined criteria. A “worst score counts” system was applied,whereby the worst score awarded for any of the given checklistitems was taken as the overall score for the methodologicalquality of the study.21 Studies with ratings of “good” or“excellent” were considered to be of high quality, and studieswith ratings of “poor” or “fair” were considered to be of lowquality. It was anticipated that older studies would not consis-tently adhere to the reporting requirements of the COSMINchecklist, given the relative newness of the guidelines and theworst score scoring structure of the checklist. Therefore, boththe Terwee et al checklist and the COSMIN checklist were usedto provide a more comprehensive review of study and mea-surement tool quality.22

Results

The study selection process for both searches is shown in thePreferred Reporting Items for Systematic reviews and Meta-Analyses flowchart (fig 1).

Part 1: Identification of upper limb measurementtools

Eighteen measures were identified (table 1).23-38 Agreement be-tween raters for full-text screening was 83.4%. Interventionsevaluated included surgical techniques, drug management and theeffects of the “on”/“off” cycle, physiotherapy, exercise, andoccupational therapy. Most studies included fewer than 25 par-ticipants and demonstrated disease durations of 5 or more years.Disease severity ranged from I to V on the Hoehn andYahr scale.39

Seven identified tools were developed specifically for theevaluation of PD or other movement disorders. These measures

Table 1 Characteristics and clinical utility of the identified measurement tools

Measurement Tool

Times

Reported Scoring

Clinical Utility

Time to

Administer

(min)

No. of

Test Items

Approximate

Cost (US $) Equipment

PD and movement disorderespecific

scales

Clinical Rating Scale for Tremor

(items 5, 6, 11e15)235 Ordinal scale 0e4 10e15 8 <25 Pen, paper, plastic cups

CAPIT upper limb timed tests24 10 Time to complete set

number of repetitions

10 3 <25 Board, 2 markers, 30cm apart

CAPSIT-PD upper limb timed test25 1 Repetitions in 20s 5 1 <25 Board, 2 markers, 30cm apart

Modified Bradykinesia Rating Scale26 1 Ordinal scale 0e4. Rate

speed, amplitude, rhythm

5 1 0 None required

MDS-UPDRS (items 3.3-6; 3.15-8)27 2 Ordinal scale 0e4 5 8 0 None required

UPDRS (Item 8: Handwriting)28 2 Ordinal scale 0e4 <5 1 0 None required

(Upper limb items 20e25)28 31 Ordinal scale 0e4 5 6 0 None required

Webster (Bradykinesia item)29 1 Ordinal scale 0e3 <5 1 0 Pen, paper

Generic measures and tools developed

for conditions other than PD

Action Research Arm Test30 1 Ordinal scale 0e3 8e30 19 495 (Salia Rehab) Wooden blocks, cricket ball,

stone, jug and glass, tube, washer

and bolt, ball bearing, marble

Box and Block Test31 1 Number of blocks

moved in 1min

5 1 230 Wooden box divided in 2 and 150

wooden cubes

Coin Rotation Task32/Test33 3 Time for specified

number of rotations

<5 1 5¢ Small coin (US ¢5)

DASH34 1 Ordinal scale 1e5 15þ 30 e Self-report questionnaire

Fugl-Meyer Assessment

(Upper limb section)351 Ordinal scale 1e3 15e20 33 <50 Reflex hammer, paper, pencil, plastic mug,

tennis ball

Grooved Pegboard Testa 3 Time to insert pegs 5 1 95 Board with 25 holes and pegs that are

rotated to match the holes

Writing task using Wacom graphics

tabletb and Scriptalyzer software36,c1 Kinematic analysis of task 5 1 500þ Graphics tablet and pen, software license

Minnesota Manual Dexterity Testa 1 Time to place disks;

time to turn disks

20 (4 trials) 2 200 Board with 60 large holes, 60 cylinders

Motor Performance Series37,a,d 4 Time to complete tasks

or accuracy

15e20 6 1900 (for 50

administrations)

Computerized test battery

Nine Hole Peg Test38 3 Time to insert and

remove pegs

10 2 80 (Jamar Rolyan) Pegboard with 9 pegs

Purdue Pegboard Testa 21 Number of pegs inserted

in 30s or constructions

in 1min

5 (1 trial

administration)

4 125 Pegboard, metal pins, washers, collars

Abbreviations: CAPSIT-PD, Core Assessment Program for Surgical Interventional Therapies in Parkinson’s disease; MDS-UPDRS, Movement Disorders Societyesponsored revision of the UPDRS.

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Upper limb measures in Parkinson disease 5

were mostly global scales containing upper limb items, and in-dividual and combined upper limb scores were reported. Com-bined scores for the Unified Parkinson’s Disease Rating Scale(UPDRS)28 included an “upper limb bradykinesia score” (sum ofitems 23, 24, and 25)40 and an “upper extremity score” (sum ofupper limb scores for items 20-22 and scores for items 23e25).41

Several reported timed upper limb tests were excluded becauseequipment or software was either not fully described or notcommercially available, or details of the test protocol were notincluded in the study. Seven generic manual dexterity tests wereidentified. One PD-specific upper limb impairment measure, theModified Bradykinesia Rating Scale,26 was reported. Noperformance-based measure developed to evaluate upper limbfunctional tasks in people with PD was identified. Three meas-uresdthe Fugl-Meyer Assessment,35 the Action Research ArmTest,30 and the Disabilities of the Arm, Shoulder and Hand(DASH) questionnaire34dwere developed specifically for otherclinical populations.

Part 2: Measurement properties of upper limb tools

The 18 included studies investigated the measurement propertiesof 10 measures (table 2).26,42-58 No studies of measurementproperties in populations with PD were identified for the Box andBlock Test, Clinical Rating Scale for Tremor, Coin Rotation Task,DASH, Fugl-Meyer Assessment, Movement Disorders Society-sponsored revision of the UPDRS upper limb items, MinnesotaManual Dexterity Test,a or Scriptalyzer writing task.c Studysample sizes ranged from 12 to 411, and mean disease duration formost samples was >5 years. Agreement between reviewers was77.3% for screening of titles and abstracts and 80% for full-textscreening of the studies.

Reliability was investigated in 9 measurement tools,construct validity in 4 measures, and responsiveness in only 1measure. COSMIN scores for study methodology with adescription of the “worst score” items are given in table 2.Percentage agreement for reviewers was 79.1% for the COSMINscoring and 88.8% for the Terwee rating, with consensus ach-ieved after discussion. One reliability study with high-qualitymethodology was identified,50 and there were no high-qualitystudies of validity or responsiveness. Several “positive”Terwee measurement tool ratings were given in reliabilitystudies of low methodological quality. In validity and respon-siveness studies, lack of detailed reporting frequently contrib-uted to ratings of poor study methodology and “indeterminate”measurement tool quality.

There was moderate support for the interrater and intraraterreliability of items 20, 21, and 23 to 25 of the UPDRS in a high-quality study that received mostly positive Terwee ratings.50

Positive ratings for the “Pronation/supination” and “Movementbetween two points” Core Assessment Program for IntracerebralTransplantations (CAPIT)/Core Assessment Program for SurgicalInterventional Therapies in Parkinson’s disease tests, Motor Per-formance Series test battery, and unimanual subtests of the PurduePegboard Test43 were also given in fair quality studies of test-retest reliability.

Investigations of the construct validity of CAPIT upper limbtests,54 Grooved Pegboard Test,55,56,a Modified BradykinesiaRating Scale,26 and Motor Performance Series57,a,d with lowmethodological quality ratings and indeterminate measurementtool ratings found moderate to strong correlations between thesemeasures and disease severity. These studies, however, scored

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poorly on both sets of quality rating because they failed to meetthe criteria for hypothesis formulation or detailed reportingrequired by the 2 checklists. The study of responsiveness of theUPDRS58 did not meet the checklist standards for good method-ology or positive measurement tool quality.

ICF LinkingWe linked each measurement tool to categories of the ICFframework12 (tables 3 and 4), with 79% agreement between the 2reviewers. The content of PD-specific measures mostly linked toimpairment categories specific to the PD disease process. In themain, generic measures linked to activity limitations contained incategory d440 Fine hand use. All timed tests were classified asbradykinesia tests (Coordination of complex voluntary move-ments). A single measure, DASH,34 was linked to participationrestrictions. Two measures developed for other clinical pop-ulations, the Fugl-Meyer Assessment35 and DASH,34 were linkedto impairments not typically associated with PD, namely, theStretch motor reflex and Touch function.

Clinical utilityInformation relating to the clinical utility of each measurement toolis included in table 1. Most assessments could be completed within10 minutes and cost <US $100 to purchase. PD-specific scalesused little equipment, and only the UPRDS and the MovementDisorders Societyesponsored revision of the UPDRS requiredadministrator training. Multifaceted test batteries took longer toadminister, and commercially produced tests were more costly.

Discussion

This is the first comprehensive review of upper limb measurementtools described in PD, and their measurement properties. Theidentified tools included those designed specifically for PD ormovement disorder evaluation, generic upper limb measures, andtools developed for other disease populations. PD-specific mea-sures were generally linked to ICF impairment categories, whereasgeneric measures and tools developed for other conditions eval-uated a range of impairments and activity limitations. Mostimpairment measures required little equipment and were quick toadminister. Higher equipment costs were associated withcommercially produced tests. Measurement properties were re-ported for 10 identified tools used to evaluate the upper limb inpeople with PD. One high-quality study supported the interraterand intrarater reliability of select UPDRS upper limb items eval-uating PD-specific impairments. Fair evidence was found to sup-port the test-retest reliability of the Purdue Pegboard Test,43,a

Motor Performance Series,44 and individual CAPIT/CoreAssessment Program for Surgical Interventional Therapies inParkinson’s disease timed tests.43 No high-quality studies inves-tigated validity or responsiveness in any of the measures, and nomeasurement tool was given a positive rating for either of theseproperties. Further high-quality research to establish the mea-surement properties of tools used to evaluate the upper limb inpeople with PD is needed to inform measurement tool selectionfor clinical and research purposes.

Impairments including tremor, rigidity, and bradykinesia werecommonly quantified using individual or combined upper limbscores from PD-specific scales such as the UPDRS. There wasevidence for the reliability of individual UPDRS items and CAPITand Core Assessment Program for Surgical Interventional Ther-apies in Parkinson’s disease upper limb tests but a lack of support

Table 2 Measurement properties of the identified measurement tools

Measurement Tool

Measurement Property

Evaluated n

Years of PD:

Mean � SD Results

Quality

Rating*COSMIN

Score COSMIN “Worst Score” Itemy

Reliability

Action Research

Arm Test

Test-retest42 20 ND ICCZ.99 ? Poor Sample size

CAPIT/CAPSIT-PD Test-retest43 34 14.1�6.7 Contralateral/ipsilateral to surgery Pronation-supination:

ICCs “off”Z.85/.26; “on”Z.78/.70; Movement between

2 points: ICCs “off”Z.54/.53; “on”Z.83/.78

þ “on”

� “off”

Fair Sample size

Modified Bradykinesia

Rating Scale

Intrarater26 100 9.5�5.6 Speed/Amplitude/Rhythm:

Finger taps rZ.61/.79/.72; Hand grasps rZ.56/.83/.73;

Pronation-supination rZ.66/.79/.79

? Poor Kappa calculated

Interrater26 100 9.5�5.6 Speed/Amplitude/Rhythm:

Finger taps rZ.51/.77/.69;

Hand grasps rZ.56/.85/.79

Pronation-supination rZ.70/.81/.78

? Poor Kappa calculated

Motor Performance

Series

Test-retest44 114 6.1�4.9 ICCs RightZ.88; LeftZ.87. Some subtest results <0.70 þ Fair Similar test conditions

Nine Hole Peg Test Test-retest,

Measurement error45262 6.2�4.8 Dominant hand ICCZ.88; SEMZ1.02s; MDCZ2.60s;

Nondominant hand ICCZ.91; SEMZ.82s; MDCZ1.30s

? Poor Independent administrations;

appropriate time interval

Intrarater46 12 6.8�4.4 ICCZ.99 (Face to face and telerehabilitation) þ Poor Sample size

Interrater46 12 6.8�4.4 ICC�.99 þ Poor Sample size

Purdue Pegboard Test Test-retest43 34 14.1�6.7 Contralateral/ipsilateral to surgery: ICCs “off”Z.71/.79;

“on”Z.87/.89

þ Fair Sample size

UPDRS Item 8 Interrater47 40 4.96�4.36 kZ.71 þ Fair Sample size

Interrater48 392 0.7�0.8 kwZ.49 (Participant/investigator) � Fair Appropriate time interval;

independent administrations

UPDRS Item 20 Test-retest49 400 1.0�1.2 kwZ.75 þ Fair Stability between tests;

appropriate time interval

Intrarater50 50 11.4�7.4 kw>0.70 þ Good Weighted kappa calculated

Interrater50 50 11.4�7.4 kw>.64e.88 � Good Weighted kappa calculated

UPDRS Item 21 Test-retest43 34 14.1�6.7 “off” ICCZ.77; “on” ICCZ.63 � Poor Kappa calculated

Test-retest49 400 1.0�1.2 kwZ.61 � Fair

Intrarater50 50 11.4�7.4 kwZ.51e.91 � Good

Interrater50 50 11.4�7.4 kwZ.45e.75 � Good

Interrater47 40 4.96�4.36 KZ.48 � Fair

Interrater51 24 5.6�4.5 ICCZ.67 � Poor Sample size; kappa calculated

UPDRS Item 22 Test-retest49 400 1.0�1.2 kw<.70 � Fair

UPDRS Item 23 Test-retest49 400 1.0�1.2 kw<.70 � Fair

Intrarater50 50 11.4�7.4 kw>.70 þ Good

Interrater50 50 11.4�7.4 kw>.70 þ Good

Interrater47 40 4.96�4.36 KZ.47 � Fair

Interrater51 24 5.6�4.5 ICCZ0.7 þ Poor

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Table 2 (continued )

Measurement Tool

Measurement Property

Evaluated n

Years of PD:

Mean � SD Results

Quality

Rating*COSMIN

Score COSMIN “Worst Score” Itemy

UPDRS Item 24 Test-retest49 400 1.0�1.2 kw<.70 � Fair

Intrarater50 50 11.4�7.4 kw>.70 þ Good

Interrater50 50 11.4�7.4 kwZ.66e.85 � Good

Interrater47 40 4.96�4.36 KZ.60 � Fair

Interrater51 24 5.6�4.5 ICCZ.75 þ Poor

UPDRS Item 25 Test-retest49 400 1.0�1.2 kw<.70 � Fair

Intrarater50 50 11.4�7.4 kwZ.60e.88 � Good

Interrater50 50 11.4�7.4 kwZ.63e.83 � Good

Interrater47 40 4.96�4.36 KZ.60 � Fair

Interrater51 24 5.6�4.5 ICCZ.89 þ Poor

Webster (bradykinesia) Interrater52 48 6.0�4.2 kZ.73 � Fair Sample size

Interrater53 48 ND kZ.43 � Fair Sample size

Validity or responsiveness

CAPIT timed tests Construct validity54 33 10.5�4.2 Correlation with UPDRS Motor: Pronation/supination

“off” rZ.64; “on” rZ.48; Finger dexterity “off”

rZ.68; “on” rZ.67

? Poor Measurement properties of

comparator instruments

Grooved Pegboard Test Construct validity55 28 3.0�3.6 Correlation between pegboard scores (least affected

arm) and least dennervated striatum rZ.69

? Poor Sample size

Construct validity56 191 ND Correlation with UPDRS III MAH rZ0.6; LAH rZ.59;

UPDRS sum 23e25 MAH rZ.37; LAH rZ.43

? Fair Hypotheses formulated

Modified Bradykinesia

Rating Scale

Construct validity26 100 9.5�5.6 Correlation with kinematic variables: speed and

amplitude r>.70; rhythm<.70

? Poor Hypotheses formulated;

measurement properties of

comparator instruments

Motor Performance Series Construct validity57 411 ND Correlation with Webster scale. Plugging Spearman

rZ.51; Fast tapping Spearman rZ.02

? Poor Measurement properties of

comparator instruments

UPDRS II and III Responsiveness58 376 ND Item 8 SRMZ�.50; (Right/Left)

Item 20 SRMZ�.47/.45

Item 21 SRMZ�.16/�.09

Item 22 SRMZ�.39/�.38

Item 23 SRMZ�.60/�.48

Item 24 SRMZ�.55/�.42

Item 25 SRMZ�.56/�.47

? Poor Hypotheses formulated

Abbreviations: ICC, intraclass correlation coefficient; k, kappa statistic; kw, weighted kappa; LAH, less affected hand; MAH, more affected hand; MDC, minimal detectable change; ND, not defined; SRM,

standardized response mean.

* Quality assessment: (þ) positive rating, quality criteria have been met; (�) negative rating, quality criteria have not been met; (?) indeterminate rating due to doubtful design or method; (�) score

includes mixed results with some positive and negative ratings. For a þ rating: ICC or K values of >.70 are necessary for reliability studies; >75% of stated “a priori” hypotheses must be met for construct

validity studies; and for responsiveness studies, the smallest detectable change is less than the minimal important change, OR the minimal important change is outside the limits of agreement OR the

responsiveness ratio is >1.96 OR the area under the curve is >.70.y COSMIN “worst score item”: Indicates COSMIN checklist item responsible for “worst score.”

Upper

limbmeasu

resin

Parkin

sondisease

7

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Table 3 Linking of identified measurement tools to the ICF “Body functions” classification

Measurement

Tool

Categories

Covered

(n)

Body Functions

b126

Temperament

and

Personality

Factors

b134

Sleep

Functions

b265

Touch

Function

b280

Sensation

of Pain

b710

Mobility

of Joints

Functions

b730 Muscle Power

Functions

b735

Muscle

Tone

Functions

b750

Motor

Reflex

Functions

b760 Control of

Voluntary Movement

Functions

b765

Involuntary

Movement

Functions

b780

Sensations

Related to

Muscles

and

Movement

Functions

b2801

Pain in

Body

Part

b7101

Mobility

of

Several

Joints

b7300

Power of

Isolated

Muscles

and Muscle

Groups

b7301

Power of

Muscles

of One

Limb

b7356

Tone

of All

Muscles

of the

Body

b7500

Stretch

Motor

Reflex

b7600

Control

of Simple

Voluntary

Movements

b7601

Control of

Complex

Voluntary

Movements

b7602

Coordination

of Complex

Voluntary

Movements

b7651

Tremor

b7800

Sensation

of Muscle

Stiffness

PD or MD-specific

measures

CAPIT 1 OCAPSIT-PD 1 OCRST items 5,

6, 11e15

2 O O

UPDRS 8, 20e25 3 O O OMDS-UPDRS

3.3e6; 3.15e8

3 O O O

Webster

bradykinesia

1 O

Measures not

specific to PD

ARAT 2 O OBBT 1 OCRT 1 ODASH 9 O O O O O O O O OFMA 6 O O O O O OGPT 1 OWriting task 1 OMMDT 1 OMPS 2 O ONHPT 1 OPPT 1 O

Abbreviations: ARAT, Action Research Arm Test; BBT, Box and Block Test; CAPSIT-PD, Core Assessment Program for Surgical Interventional Therapies in Parkinson’s disease; CRST, Clinical Rating Scale for

Tremor; CRT, Coin Rotation Test; FMA, Fugl-Meyer Assessment; GPT, Grooved Pegboard Test; MD, movement disorders; MDS-UPDRS, Movement Disorders Societyesponsored revision of the UPDRS; MMDT,

Minnesota Manual Dexterity Test; MPS, Motor Performance Series; NHPT, Nine Hole Peg Test; PPT, Purdue Pegboard Test.

8E.L.

Proudet

al

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Table 4 ICF Linking to ICF Activities and Participation component

Measurement

Tool

Categories

Covered (n)

Activities and Participation

d210

Undertaking

a Single

Task

d230

Carrying

Out Daily

Routine

d430 Lifting and

Carrying Objects d440 Fine hand use d445 Hand and Arm Use

d470

Using

Transport

d 473

Driving

d510

Washing

Oneself

d520

Caring

for Body

Parts

d540

Dressing

d550

Eating

d4300

Lifting

d4301

Carrying

in the

Hands

d4305

Putting

Down

Objects

d4400

Picking

Up

d4401

Grasping

d4402

Manipulating

d4452

Reaching

d4453

Turning or

Twisting

the Hands

or Arms

d4454

Throwing

d5100

Washing

Body

Parts

d5202

Caring

for Hair

d5400

Putting

on

Clothes

CRST (5, 6,

11e15)

1 O

ARAT 6 O O O O O OBBT 2 O OCRT 1 ODASH* 14 O O O O O O O O O O O O O OFMA 2 O OGPT 3 O O OMMDT 4 O O O OMPS 4 O O O ONHPT 3 O O OPPT 3 O O O

Abbreviations: ARAT, Action Research Arm Test; BBT, Box and Block Test; CRST, Clinical Rating Scale for Tremor; CRT, Coin Rotation Test; FMA, Fugl-Meyer Assessment; GPT, Grooved Pegboard Test; MMDT,

Minnesota Manual Dexterity Test; MPS, Motor Performance Series; NHPT, Nine Hole Peg Test; PPT, Purdue Pegboard Test.

* Activity and participation items included only in DASH: d630 Preparing meals, d640 Doing housework, d6402 Cleaning living areas, d650 Caring for household objects, d6501 Maintaining dwelling and

furnishings, d6505 Taking care of plants indoors and outdoors, d7500 Informal relationships with friends, d7501 Informal relationships with neighbors, d7504 Informal relationships with peers, d760 Family

relationship, d7702 Sexual relationship, d8451 Maintaining a job, d850 Remunerative employment, d855 Nonremunerative employment, d9200 Play, d9201 Sports, d9202 Arts and Culture, d9203 Crafts, d9204

Hobbies, d9205 Socializing.

Upper

limbmeasu

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sondisease

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10 E.L. Proud et al

for their validity and responsiveness. Although individual test itemscores are often used to describe the presence or severity of im-pairments, there was little evidence to support their use in eval-uating treatment effects. Developing technologies includingcomputer software programs, digitizing tablets, and smartphoneapplications are now able to provide detailed, objective informa-tion about PD-specific impairments. These sophisticated mea-surement techniques may prove to be more responsive thansubjective scales such as the UPDRS to change59; however, theirmeasurement properties are yet to be established in this clin-ical group.

Few activity measures were the subject of reliability or validitytesting in PD, and no studies of responsiveness were found. Therewas, however, limited support for the test-retest reliability of thePurdue Pegboard Test. Although no high-quality evidence wasfound for the validity or responsiveness of the “pegboard” tests,moderately strong relations were observed between diseaseseverity in PD and pegboard test scores,45,56 suggesting that thesemeasurement tools may be worthy of further investigation.Pegboard tests quantify fine hand coordination,56 which is a keyfeature of upper limb disability in this clinical population.6

Changes in test performance noted with disease progression,45

altered medication state,60 and after surgical intervention61 indi-cate that these measures may also be responsive to change in thisclinical group. On the basis of current evidence, the PurduePegboard Test could be considered for quantifying hand functionin PD but will require further evaluation to support its validity andresponsiveness and to facilitate the interpretation of test results.

In accordance with the COSMIN administration guidelines, a“worst score counts” scoring system was used to rate each study’smeasurement properties on the basis of the quality of the studymethodology. Because many of the included studies were writtenbefore the development of the COSMIN checklist, quality ratingsmay reflect not only methodological flaws but also reportingstandards at the time of publication. For example, in studiesinvestigating construct validity, it is expected that hypothesesclearly describing the expected relations between measurementvariables are stated “a priori.”22 In the identified studies, hy-potheses were not clearly articulated and descriptions of theconstructs being measured and tools under investigation lackeddetail. In addition to providing a means of evaluating studies ofmeasurement properties, the COSMIN checklist provides criteriafor high-quality study design21 and could assist in the planningand reporting of future studies of this type.

Three measures developed for clinical populations other thanPD were identified in this review: the Fugl-Meyer Assessmentand Action Research Arm test for individuals after stroke30,35 andthe DASH for people with musculoskeletal shoulder disorders.34

Measures such as these can include test items evaluatingcondition-specific impairments and functional limitations that areinappropriate or irrelevant to upper limb disability in people withPD, potentially affecting their validity and responsiveness in thisclinical group. There are few recommendations for selectingupper limb measurement tools for this patient group,6,8,9 andtherefore clinicians may use “familiar” measurement toolsdeveloped for other disease populations.10 It is important tocarefully examine the content of these tools in light of the spe-cific upper limb difficulties experienced by people with PD, suchas bradykinesia and reduced dexterity. The ICF-based contentcomparison provided here may assist in the selection of measuresthat warrant further investigation as tools for evaluating the upperlimb in people with PD.

This review highlights the lack of high-quality evidenceavailable to guide clinicians and researchers in the selection ofmeasurement tools to evaluate change in upper limb impairments,activity limitations, and associated participation restrictions inpeople with PD. Specifically, the validity and responsiveness ofmeasurement tools were poorly addressed in this clinical group.This knowledge is essential for the informed selection of mea-surement tools and the evaluation of treatment effects. BecausePD is a progressive condition and the motor presentation is het-erogeneous, the spectrum of upper limb functional deficits mayrange from mildly decreased dexterity to an inability to completeself-care tasks. It is therefore important to extend our under-standing of the measurement properties of these tools when theyare administered in different stages of the disease.

Future directions

Key areas of future research could include further examination of thereliability of a selection of measurement tools identified in this re-view. Given the daily fluctuations in motor performance that mayoccur in people with PD,62 test-retest reliability is of particularsignificance in this population. The identification of measures suit-able for the evaluation of rehabilitation programs is also a priority.Measurement tools to be investigated for their measurement prop-erties and clinical utility may be selected from those identified in thereview, or from the many other available measures of upper limbfunction. The ICF content comparison presented here may assist inidentifying measures that address priority assessment needs. Otherresearch priorities could include the development of a PD-specificmeasure to quantify the performance of upper limb daily tasks anda tool to evaluate participation restrictions with a focus on the effectof upper limb difficulties on participation in people with PD.

Study limitations

Limitations of this review included possible bias due to theexclusion of studies not published in English. A further limitationwas the inclusion of only those measures described in interventionstudies, therefore excluding measures currently in the develop-ment or evaluation phase such as the Bradykinesia AkinesiaIncoordination test.63,64

Conclusions

This review provides new information to guide clinicians and re-searchers in the selection of measurement tools for upper limb eval-uation in people with PD. It also confirms existing gaps in ourknowledge of upper limb measurement in this clinical population, inparticular the lack of evidence for the validity and responsiveness ofmeasures in current use. Without this information, it is difficult tocorrectly interpret the meaning of change in measurement aftertreatment and the effectiveness of upper limb interventions. Currentevidence supports the reliability of a small number of the identifiedmeasures, but further investigation of the validity and responsivenessof these and other upper limb tools is required to determine theirsuitability for quantifying the specificupper limbdeficits found inPD.

Suppliers

a. Lafayette Instrument Co, 3700 Sagamore Pkwy, N Lafayette,IN 47904.

b. Wacom, 1311 SE Cardinal Crt, Vancouver, WA 98683.

www.archives-pmr.org

Upper limb measures in Parkinson disease 11

c. Neuroscript LLC, 435 E Carson Dr, Tempe, AZ, 85282.d. Schuhfried GmbH, Hyrtlstrasse 45, 2340 Modling, Austria.

Keywords

Outcome assessment (health care); Parkinson disease; Physicaltherapy specialty; Psychometrics; Rehabilitation; Upper extremity

Corresponding author

Elizabeth L. Proud, MPhysio (Neuro), Department of Physio-therapy, School of Health Sciences, Level 7, Alan Gilbert Bldg,The University of Melbourne, 3010 Victoria, Australia. E-mailaddress: [email protected].

Acknowledgment

We thank Clarissa L. Martin, PhD, for her contribution to theinitial development of the systematic review protocol/procedures.

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www.archives-pmr.org

Supplemental Appendix S1 SearchStrategies Used in Scopus

Search 1: Identification of measurement tools

TITLE-ABS-KEY(parkinson* OR “parkinson* disease”) ANDTITLE-ABS-KEY(“upper limb” OR “upper extremity” OR

arm* OR hand*) ANDTITLE-ABS-KEY(measure* OR assess* OR outcome$ OR

tool$ OR instrument* OR dexterity OR disability OR ADL) AND.(LIMIT-TO(DOCTYPE, “ar”)) AND (LIMIT-TO(LAN-

GUAGE, “English”))

Search 2: Identification of measurement properties

TITLE-ABS-KEY(dyskinesia* OR hyperkinesia* OR hypo-kinesia OR tremor OR “Activities of Daily Living” OR “MuscleRigidity” OR movement OR “motor performance” OR “motoractivity” OR dexterity OR handwriting) AND TITLE-ABS-KEY(parkinson* disease) AND

TITLE-ABS-KEY(“unified parkinson’s disease rating scale”OR UPDRS OR MDS-UPDRS OR “Core Assessment Program forIntracerebral Transplantations” OR CAPIT OR CAPSIT-PD OR“Core Assessment Program for Surgical Interventional Therapiesin Parkinson’s disease” OR “Clinical rating scale for tremor” OR“modified bradykinesia rating scale” OR “Webster scale” OR“purdue peg*” OR “grooved pegboard” OR “coin rotation t*” OR“nine hole peg test” OR “9 hole peg test” OR “box and block test”OR “motor performance test” OR “Motorische Leistungsserienach Schoppe” OR “disabilities of the arm shoulder and hand” ORDASH OR “Fugl-Meyer assessment” OR FMA OR “actionresearch arm test” OR ARAT OR “minnesota manual dexterity”OR Scriptalyzer OR “graphics tablet” OR “digitizing tablet”)AND

TITLE-ABS-KEY(psychometr* OR “Validation Studies” OR“Comparative Study” OR “observer variation” OR “reproduc-ibility of results” OR “discriminant analysis” OR clin?metr* ORvalid* OR person?metric OR reliab* OR unidimensional* ORdiscriminability OR appropriateness OR precision OR interpret-ability OR acceptability OR practicability OR “floor effect” OR“ceiling effect” OR “standardised response mean” OR SRM OR“Guyatt’s responsiveness statistic” OR “effect size” OR reprodu-cib* OR unreliab* OR coefficient OR homogeneity OR

homogeneous OR “internal consistency” OR “cronbach* alpha*”OR “item correlation*” OR “item selection*” OR “item reduc-tion*” OR agreement OR imprecision OR “precise values” OR“test retest” OR “test-retest” OR “reliab* test” OR “reliab* retest”OR stability OR interrater OR inter-rater OR intrarater OR intra-rater OR intertester OR inter-tester OR intratester OR intra-testerOR interobserver OR inter-observer OR intraobserver ORintra-observer OR intertechnician OR inter-technician OR intra-technician OR intra-technician OR interexaminer OR inter-examiner OR intraexaminer OR intra-examiner OR interassayOR inter-assay OR intraassay OR intra-assay OR interindividualOR inter-individual OR intraindividual OR intra-individual ORinterparticipant OR inter-participant OR intraparticipant ORintra-participant OR kappa OR kappa’s OR kappas OR repeatab*OR “replicab* measure*” OR “replicab* findings” OR “repli-cab* result*” OR “replicab* test*” OR “repeated measure*” OR“repeated findings” OR “repeated result*” OR “repeated test*”OR generaliza* OR generalisa* OR concordance OR “intraclasscorrelation*” OR discriminative OR “known group” OR “factoranalysis” OR “factor analyses” OR dimension* OR subscale*OR “multitrait scaling analys*” OR “item discriminant” OR“interscale correlation*” OR error* OR “individual variability”OR “variability analys*” OR “variability values” OR “uncer-tainty measurement” OR “uncertainty measuring” OR “standarderror of measurement” OR sensitiv* OR responsive* OR “min-imal* detectable change” OR MDC OR “minimal* clinical*important* difference” OR “minimal* clinical* significant dif-ference” OR “minimal* clinical* detectable* difference” OR“minimal* clinical* important* change” OR “minimal* clinical*significant change” OR “minimal* clinical* detectable* change”OR “clinical* important* difference” OR “clinical* significantdifference” OR “clinical* detectable* difference” OR “clinical*important* change” OR “clinical* significant change” OR“clinical* detectable* change” OR “small detectable change”OR “small real change” OR “small detectable difference” OR“small real difference” OR “clinical* important* difference” OR“clinical* significant difference” OR “clinical* detectable* dif-ference” OR “clinical* important* change” OR “meaningfulchange” OR “Item response model” OR IRT OR rasch OR“Differential item functioning” OR DIF OR “computer adaptivetesting” OR “item bank” OR “cross-cultural equivalence”) AND(LIMIT-TO(SRCTYPE, “j”)) AND (LIMIT-TO(LANGUAGE,“English”))

Upper limb measures in Parkinson disease 12.e1

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