Fatigue in Child Chronic Health Conditions: A Systematic ... fatigue assessment instruments used in...

Fatigue in Child Chronic HealthConditions: A Systematic Review ofAssessment InstrumentsAlison Crichton, DPsycha,b,c, Sarah Knight, PhDa,b,d, Ed Oakley, PhDb,d,e, Franz E. Babl, MDb,d,e, Vicki Anderson, PhDb,c,e

abstractBACKGROUND AND OBJECTIVE: Fatigue is common in chronic health conditions in childhood, associatedwith decreased quality of life and functioning, yet there are limited data to compareassessment instruments across conditions and childhood development. Our objective was todescribe fatigue assessment instruments used in children with chronic health conditions andcritically appraise the evidence for the measurement properties of identified instruments.

METHODS: Data sources included Medline, Cumulative Index to Nursing and Allied HealthLiterature, and PsycINFO (using the EBSCOhost platform). Study selection includedquantitative assessment of fatigue in children with health conditions. Data extraction was asfollows: (1) study design, participant and fatigue instruments, (2) measurement properties offatigue instruments, (3) methodological quality of included studies, and (4) synthesis of thequality of evidence across studies for the measurement properties of fatigue instruments.

RESULTS: Twenty fatigue assessment instruments were identified (12 child reports, 7 parentreports, 1 staff report), used in 89 studies. Fatigue was assessed in over 14 health conditions,most commonly in children with cancer and chronic fatigue syndrome. Evidence for themeasurement properties of instruments varied, and overall quality was low. Two fatigueinstruments demonstrated strong measurement properties for use in children with diversehealth conditions and children with cancer.

CONCLUSIONS: The review is limited to children younger than 18 years and results are specific tohealth conditions described, limiting generalizability of findings to other populations. Evidencefor the measurement properties of fatigue instruments varied according to the population inwhich instruments were used and informant. Further evidence is required for assessment offatigue in younger children, and children with particular health conditions.

aVictorian Pediatric Rehabilitation Service, Monash Children’s, Melbourne, Australia; bMurdoch Childrens Research Institute, Melbourne, Australia; cSchool of Psychological Sciences anddDepartment of Pediatrics, University of Melbourne, Melbourne, Australia; and eRoyal Children’s Hospital, Melbourne, Australia

Dr Crichton conceptualized and designed the study, carried out the literature searches, completed the initial analysis, drafted the initial manuscript, and revised themanuscript; Dr Knight assisted with the data collection instrument, screened articles for inclusion, reviewed analysis, and reviewed the manuscript; Dr Andersonsupervised data collection and critically reviewed the manuscript; Drs Oakley and Babl cosupervised data collection and reviewed the manuscript; and all authorsapproved the final manuscript as submitted.

www.pediatrics.org/cgi/doi/10.1542/peds.2014-2440

DOI: 10.1542/peds.2014-2440

Accepted for publication Dec 30, 2014

Address correspondence to Alison Crichton, DPsych, Senior Clinical Neuropsychologist, Critical Care and Neurosciences Theme, Murdoch Childrens Research Institute,Royal Children’s Hospital, Flemington Rd, Parkville, 3052, Australia. E-mail: [email protected]

PEDIATRICS (ISSN Numbers: Print, 0031-4005; Online, 1098-4275).

Copyright © 2015 by the American Academy of Pediatrics

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Fatigue refers to a subjectivelyoverwhelming sense of tiredness, lackof energy, and feeling of exhaustion.1

It is a common, independent, andnonspecific symptom identified innumerous chronic health conditionsin childhood. Consequently, it hasbeen described as 1 of the mostuniversal experiences for childrenwith chronic health conditions,2

although reported rates of fatigue varywithin and between conditions.3–7

Excessive fatigue leads to reducedquality of life,8 depression,9 andreduced school participation.10–13

Fatigue is also identified in healthychildren with normal daily functioning.Given these characteristics, it isimportant to identify accurate andreliable child-friendly measures todetect excessive fatigue.

EPIDEMIOLOGY

Excess fatigue is a common presentingproblem in clinical practice, occurringin children with cancer,14,15

neurologic conditions,16 chronicfatigue syndrome,17 posturaltachycardia syndrome,18 multiplesclerosis,6 epilepsy,19 traumatic braininjury,20 juvenile idiopathic arthritis,21

diabetes,22,23 inflammatory boweldisease,24 fibromyalgia,25,26 andobesity.27 Importantly, fatigue in theseconditions is distinguishable fromother related symptoms includingsleepiness, depression, and apathy,28

and is not ameliorated by sleep.1

Precise prevalence estimates areconstrained by issues related toassessment, terminology, andmeasurement, which are heightenedin childhood.

MEASUREMENT

Fatigue has been defined as anoverwhelming and sustained sense ofexhaustion that decreases one’scapacity for physical and mentalwork.29 Quantification of fatigueremains impeded by lack ofconsensus framework, terminology,and the subjective andmultidimensional nature of

symptoms. Three alternativeframeworks for describing subjectivefatigue are as follows: “centralfatigue” (resulting from central asopposed to peripheral nervoussystem damage and incorporatingmental and physical symptoms)30; asa “feeling state”;28 and asa combination of primary (diseasespecific) and secondary factors (suchas pain, sleep disturbance, or moodchanges).31 The subjectivity andbreadth of symptoms makequantification challenging, andresultantly assessment instrumentsfocus on the duration or severity ofsymptoms (physical, mental,affective) and other dimensions of thefatigue experience, including impacton daily functioning and exacerbatingfactors (eg, sleep habits).31–36 In thisreview, based on earlier work,37 thetheoretical construct of fatigue will becategorized as “impact of fatigue ondaily life,” “fatigue severity,” or“factors influencing fatigue.”

It is important for instruments thatassess children’s fatigue to be childspecific. It is a unique experience withits own symptomatology,presentation, and naturalhistory.38–41 Fatigue is inherentlysubjective,28 and its assessment inchildhood is challenging because theaccuracy and relevance of parent-proxy reports is questionable.42

Further, fatigue in the generalchildhood population varies throughdevelopment; it is greater in infancy,early childhood, and late adolescencethan during midchildhood, and ismore common in girls.43–45 Accurateassessment requires recognition ofthe developmental stages andreference to age and gender norms.

Although fatigue is a commonsymptom in a range of chronic healthconditions, and notable challengeswith its measurement exist, there hasbeen limited evaluation of assessmentinstruments in use with children.

TABLE 1 Data Extraction Items

Data Category Description Data Extracted

1 Study description Study aims, design, study duration, and any bias. Participantnumbers, setting, diagnostic criteria, age, gender, country,comorbidity, and diagnostic criteria used.

2 Instrument description For each study: name of instrument(s), language version,theoretical constructs, number of items, description ofdomains assessed, theoretical construct,a age, respondents,response format, and extent of active patient involvement ininstrument development.b

3 Psychometric propertiesof fatigue measures

Criteria ratingc for instrument-level measurement properties(as reported in study): (1) internal consistency, (2)reliability, (3) measurement error, (4) content validity, (5)construct validity, (6) structural validity, (7) hypothesistesting, (8) cross-cultural validity, and (9) responsiveness.

4 Study quality Ratingd of study methodological quality for each of thefollowing measurement properties reported: (1) internalconsistency, (2) reliability, (3) measurement error, (4)content validity, (5) construct validity, (6) structural validity,(7) hypothesis testing, (8) cross-cultural validity, (9)responsiveness (add * if measurement properties were notchecked, and the authors cite published measurementproperties in another article).

5 Acceptability andfeasibility

Acceptability (respondent burden, time for completion) andfeasibility (time for scoring, ease of scoringe).

6 Risk of bias Study and outcome level data: outcome data completeness,drop-out rate description, any selective outcome reporting.

a Theoretical construct of fatigue rated as impact of fatigue on daily life, fatigue severity, or factors influencing fatigue.37b INVOLVE definitions of extent of patient involvement in instrument development (“consultation,” “collaboration,” or “user-led”).136c Criteria for good measurement properties on the basis of criteria proposed by Terwee et al.55d The quality of each included study was evaluated by using the COSMIN checklist54 and rating system.57e Ease of scoring criteria were “easy,” “moderate,” or “difficult” according to Elbers et al.37

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Exceptions are reviews of measuresin children with cancer46,47 andchronic fatigue syndrome.48 It isimportant to identify the instrumentsavailable and to evaluate theirmeasurement properties. Theobjectives of this review of fatigue inchildren and adolescents with chronichealth conditions were to (1)describe participant and studycharacteristics of included studies,(2) describe and systematicallyreview available instruments used toassess fatigue, detailing thoseavailable for child (self) and parent(proxy) report, (3) interpretinstruments by usinga multidimensional framework offatigue, (4) review identifiedinstruments within a developmentalframework, and (5) summarize andsynthesize the psychometricproperties of the these instrumentsand the quality of the evidence.

METHODS

Information Sources

The search strategy was designed toretrieve literature relating tomeasures of subjective fatigue inchildren with chronic healthconditions. The systematic reviewwas conducted according to thePreferred Reporting Items forSystematic Reviews and Meta-Analyses (PRISMA).49 The EBSCOhostplatform was used to search theelectronic databases Medline,Cumulative Index to Nursing andAllied Health Literature, andPsycINFO from 1990 to August 24,2013 (updated June 12, 2014).Nonconventional documents (gray

literature) and publications,50 andsecondary references were alsochecked.

Search

The searches were limited to English-language publications. Subjectheadings and key words used werethe following: (1) fatigue, weary,weariness, exhaustion, exhausted,lacklustre or astheni*, letharg*, tired;or (2) (lack* or loss or lost) and(energy or vigor or vigor); (3) feelingand (drained or sleepy or sluggish orweak*); (4) central nervous systemdiseases, neoplasms, multiplesclerosis, congenital heart defects,anemia, chronic pain, chronic disease,cerebral palsy, cardiovasculardiseases, diabetes mellitus (type 1);chronic fatigue syndrome; and (5)questionnaires or psychological tests.

Eligibility Criteria

Population

Children and/or adolescents (#18years of age) with a health condition.

Assessment Tool

Any questionnaire designed tomeasure fatigue by using more than1 item. Fatigue subscales included inquality-of-life or other measureswere eligible for inclusion, providedthat fatigue scores could beextracted separately (ie, totalsubscale scores provided). Fatigueassessment must be subjective,quantified, and based on child reportor parent/other report, to be eligiblefor inclusion.

Outcome

Any clinical outcome.

Study Design

Interventional and observationalstudies published in peer-reviewedjournals were eligible for inclusion.

Exclusion Criteria

Review articles, opinion pieces, andsingle case studies were excluded asfollows: studies that only reportedqualitative information as a measureof fatigue (ie, professional opinion orinterview data); fatigue measuresthat used a single item, either standalone (single item visual analog scales[VASs]) or as part of anotherquestionnaire (ie, quality of life/depression/symptom checklists); andobjective measures of performancedecrement (ie, attention vigilance,muscle fatigue).

Study Selection

Titles and abstracts of all articleswere assessed for inclusion by 2independent reviewers (Drs Crichtonand Knight) and agreement checked.

Data Collection Process

A standardized data extraction formwas developed and informed bystandard processes for systematicreviews,51,52 earlier reviews in thefield,37,46–48 and incorporatingrecommendations for patient-reported outcome measureevaluation and standards forappraisal of methodologicalquality.53–55 The form wasindependently piloted in 2 randomlyselected studies by Drs Crichton andKnight. Data were extracted by DrCrichton, independently checked byDr Knight, and disagreements wereresolved by consensus withconsultation of a third reviewer(Dr Anderson; Table 1).

The data extraction and assessmentincluded information from 6 domains(Table 1). The measurementproperties evaluated in datacategories 3 and 4 in Table 1incorporate a consensus-basedtaxonomy of measurement propertiesconsidered important in patient-reported outcome measures, defined

TABLE 2 Levels of Evidence for Measurement Properties of Included Fatigue Instruments

Level of Evidence Rating Criteria

Strong +++ or 2 2 2 Consistent findings in multiple studies of good methodologicalquality or in 1 study of excellent methodological quality

Moderate ++ or 2 2 Consistent findings in multiple studies of fair methodologicalquality or in 1 study of good methodological quality

Limited + or 2 One study of fair methodological qualityConflicting +/2 Conflicting findingsUnknown ? Only studies of poor methodological quality

+, positive rating; ?, indeterminate rating; 2, negative rating.55

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by Mokkink et al56 (Table 2, p. 743).Relevant properties include thefollowing: (1) internal consistency,(2) reliability, (3) measurement error,(4) content validity, (5) constructvalidity, (6) structural validity, (7)hypothesis testing, (8) cross-culturalvalidity, and (9) responsiveness.Criterion validity is not relevant forfatigue assessment, so it wasexcluded. The 9 listed measurement

properties provided the domains fordata extraction for 2 key data sets atan individual study level: (1) ratingthe measurement property of theinstrument; (2) rating the quality ofthe study (data categories 3 and 4,Table 1; floor/ceiling effects andinterpretability not rated). Accordingto predefined criteria, eachmeasurement property was rated as“positive,” “indeterminate,” “negative,”

or “no information available.”55 Ifmeasurement properties were notchecked, but the authors citedpublished measurement properties inanother article, this was recorded andthe above ratings were maintained.

The methodological quality ofincluded studies were evaluated byusing the consensus-based standardsfor the selection of healthmeasurement instruments (COSMIN)checklist and 4-point rating scale.57

The COSMIN checklist contains 114data points (12 boxes, 9 assessingmeasurement properties), relating todesign requirements and preferredstatistical methods and consensus onstatistical values consideredadequate. Of these, 91 data points(8 boxes) were relevant to the currentreview: internal consistency (11),reliability (14), measurement error(11), content validity (5), structuralvalidity (7), hypothesis testing (10),cross-cultural validity (15), andresponsiveness (18). Each item wasscored on a 4-point rating scale (ie,“poor,” “fair,” “good,” or “excellent,”and “not applicable” if omitted). Foreach measurement property, the finalrating was determined by the lowestrating of any of the items within thebox. If more than 1 instrument wasincluded in a study (eg, parent andchild versions), the samemeasurement property was assessedfor each instrument.

Data Synthesis

Study level data were synthesized toprovide summary information foreach fatigue instrument. Informationacross studies was summarized todescribe identified instruments andthe populations to whom instrumentswere administered. Second, asguidelines for pooling ofmeasurement properties are stillunder development, best evidencesynthesis was performed integrating:(1) the rating for the instrument’smeasurement property acrossstudies55 and (2) the COSMIN ratingfor the methodological quality of thestudies.57 For each fatigue

FIGURE 1PRISMA flow diagram of study identification and selection. a The sum of the included instruments ishigher than n = 89 because some studies evaluate more than 1 measure.


instrument, the consistency acrossstudies was reviewed for eachmeasurement property. The level ofevidence for the measurementproperties of instruments acrossstudies was rated by using predefinedcriteria of positive, indeterminate, ornegative, accompanied by levels ofevidence, as proposed by theCochrane Back Review Group(Table 1),52 detailed in Table 2.

RESULTS

Figure 1 illustrates the flow of articleselection. A total of 89 studiesmeasuring fatigue by using 20instruments were included in dataextraction and synthesis.4,6,7,11,12,17,20,23–25,27,33,36,38,58–132 One studywas not included in data synthesis formeasurement properties becauseinsufficient information was availablefor data extraction.97

Patient and Study Characteristics

Most reviewed studies were cross-sectional (55; 62%), with a mediansample size of 69 subjects(6–3890). Instruments were used toassess fatigue in children witha number of health conditions(grouped into 14 diagnosticcategories). The most frequentlystudied patient groups werehematology/oncology diagnoses(37; 42%) and chronic fatiguesyndrome (17; 19%; Fig 2).

Identified Instruments

Twenty fatigue instruments wereidentified (Fig 3). The PediatricQuality of Life Inventory (PedsQL)Multidimensional Fatigue Scale (MFS)parent and child report was the mostcommonly used, cited in 26 (29%)and 20 (22%) studies,respectively.20,23–25,27,36,64,65,70,72,74,84,86,93,94,98,100,103,111,115,

116,118,125,126,129,131 The fatiguescales (Fatigue Scale-Child [FS-C];Fatigue Scale-Adolescent [FS-A], andthe Fatigue Scale-Parent [FS-P]) werethe second most commoninstruments to be cited in 18 (20%),16 (18%), and 12 (13%) studies,

respectively.38,61,67,68,75,76,78–83,85,95,107–109,113,128,132

Most (69; 77%) studies assessedfatigue by using 1 or more of 12identified child report instruments,whereas 48 (54%) of includedstudies assessed fatigue by using 1 ormore of 7 identified parent reportinstruments. Five studies (6%)included staff (“other”) report, using1 instrument. Insufficient informationwas available for 1 parent reportquestionnaire.97 Descriptive

information on the child andparent (and other) reportinstruments to assess fatigue arefound in Tables 3 and 4,respectively. See Table 5 forrespondent types within studies.

Respondent type varied across thediagnostic groups of participants; instudies of children with chronicfatigue syndrome, child reportinstruments were exclusively used,whereas studies of children withhematology/oncology diagnoses were

FIGURE 2Child health conditions of patient group in included studies, n (%).

FIGURE 3Fatigue instruments used across identified studies, n (%).


TABLE3

Characteristicsof

Included

Child

Report

Fatigue

Instruments

Measure

Construct

Assessed

aOtherConstructs

Participant

AgeRange

Healthy

Controls

AgeRange

(n=studies)

RecallPeriod

Dimension

(n=items,

range)

Response

Options

Health

Conditions

Assessed

Administration

Burden

Timeto

Complete

Ease

ofScoring

PedsQL

MFS

(child)

Severityof

fatigue;

impact

offatigue

Sleep/tiredness;

cognitive

function

5–7;8–18

5–18

(9)

1mo

Generalfatigue

(6);

sleep/rest

fatigue

(6);cognitive

fatigue

(6);totalfatigue

(18,

0–100)

5-pointLikert

(ages

8–18);4-point

Likert(ages5–7)

Hematology/

oncology;

neurologic

disease;

rheumatological

disease;obesity;

chronicpain;

braininjury;

heartdiseases

(congenital);

diabetes,type1;

gastroenterology

Administrator

required

toread

and

complete

child

report

5–7y

5min

Difficult

FS-C

Severityof

fatigue;

impact

offatigue

Cognitive

andphysical

weariness;added

effortandassistance

needed

todo

usual

activities;needing

rest

andfeeling

angry;avoiding

social

activities

7–12

7–12

(2)

24hor

1wk

or4wk

Intensity

(14;0–70);

frequency(14;0–14);

totalfatigue

(10or

14,0–70)

5-pointLikert

yes/

no,then4-point

Likert

Hematology/

oncology

Ableto

becompleted

by6-y-olds

3–10

min

Easy

FS-A

Severityof

fatigue;

impact

offatigue

Sleep/tiredness;m

ood;

cognitive

function;

physical

activity;

school

performance;

withdraw

al

13–18

13–18

(1)

24hor

1wk

or4wk

Intensity

(14;0–56

or14–70)

5-pointor

4-point

Likert

Hematology/

oncology

Notspecified

2–4min;4–5

min

Easy

CIS-20

Severityof

fatigue;

impact

offatigue

Subjectiveexperience

offatigue;

concentration;

motivation;

physical

activity

10–18

10–17

(5)

2wk

Subjectivefatigue

(8);

concentration(5);

motivation(4);

physical

activity

(3);

total(20;20–140)

7-pointLikert

Neurologic/CFS

Notspecified

Notspecified

Moderate

Chalder

Fatigue

Scale

Severityof

fatigue

Physical;m

entalfatigue

5–19

10–19

(3)

Physical(7);mental(4);

totalfatigue

(11

or14)

3-or

4-item

Likert

Hematology/

oncology

CFS

Notspecified

Notspecified

Easy

FSS

Severityof

fatigue;

impact

offatigue

Notspecified

8–18

b8–18

(2)

1wk

Totalfatigue

(9;1–7

average)

7-pointLikert

Hematology/

oncology;

neurologic

disease

Notspecified

Notspecified

Easy

PedsQL

3.0CP

Module

(child)

Notspecified

Notspecified

5–18

5–18

(1)

1mo

Totalfatigue

(4;0–100)

assubdom

aintotal

ofscalewith

7domains

5-pointLikert

(ages

8–18);4-point

Likert(ages5–7)

Neurom

otor

Administrator

required

toread

and

complete

child

report

5–7y

30min

including

consent

Difficult


TABLE3

Continued

Measure

Construct

Assessed

aOtherConstructs

Participant

AgeRange

Healthy

Controls

AgeRange

(n=studies)

RecallPeriod

Dimension

(n=items,

range)

Response

Options

Health

Conditions

Assessed

Administration

Burden

Timeto

Complete

Ease

ofScoring

VAS

Severityof

fatigue

Notspecified

8–18

None

Presentstate

Totalfatigue

(1or

6;0–4or

0–100mm)

Visual

analog

Hematology/

oncology;

musculoskeletal

Notspecified

Notspecified

Moderate

POMS

Severityof

fatigue

Notspecified

12–17

None

1wkor

4wk

Totalfatigue

(5or

15;

notspecified)as

subdom

aintotalof

scalewith

6domains

5-pointLikert

Hematology/

oncology;

neurologic

disease

Notspecified

Notspecified

Easy

PROM

IS(child)

Severityof

fatigue;

impact

offatigue

Tiredness,lack

ofenergy,impact

onactivities

8–17

8–17

(1)

1wk

Fatigue

(34/10,short

form

/15,CAT)

assubdom

aintotalof

scalewith

6domains;fatigue

item

bank:tiredness

(39);energy(14);

total(39;m

eanof

50andSD

of10)

5-pointLikert

Obesity;diabetes,

type

1;other

chronichealth

Computer

literacy,

computer,

screen,and

mouse

access

25min

younger

children,15

or32

min

adolescents

Difficult

Q-sort

task

Severityof

fatigue;

impact

offatigue

Energy

andrelated

capacityforphysical

functioning;fatigue

relatedto

psychosocial

functioning;fatigue

uniquelyassociated

with

anem

iaand/or

treatm

ent

12–18

None

1wk

Totalfatigue

(37;factor

array22to

+2)

Rank

order24to

+4Hematology/

oncology

Notspecified

Notspecified

Difficult

PedsQL

3.0

ESRD

Module

(child)

Severityof

fatigue;

impact

offatigue

Notspecified

5–18

5–18

(1)

1mo

Generalfatigue

(4;

0–100)

5-pointLikert

Renaldisease

Notspecified

Notspecified

Difficult

CAT,computerizedadaptivetesting;CFS,chronicfatigue

syndrome.

aBasedon

earlierworkby

Elbers

etal,37

thetheoretical

constructof

fatigue

was

categorizedas

impact

offatigue

ondaily

life,fatigue

severity,orfactorsinfluencingfatigue.

bGorettiet

al4included

childrenyoungerthan

17y,11

mo.Agerangewas

notspecified.


TABLE4

Characteristicsof

Included

Parent

andOtherReport

Fatigue

Instruments

Measure

Construct

Assessed

aOtherConstructs

Participant

AgeRange

Healthy

Controls

AgeRange

(n=studies)

Recall

Period

Dimension

(n=Items,

Range)

Response

Options

Health

Conditions

Assessed

Administration

Burden

Timeto

Complete

Ease

ofScoring

PedsQL

MFS

(parent)

Severityof

fatigue;

impact

offatigue

Sleepand

tiredness;

cognitive

function

2–18

2–18

(11)

1mo

Generalfatigue

(6);sleep/

restfatigue

(6);cognitive

fatigue

(6)(total

=18;

0–100)

5-pointLikert

Hematology/oncology;

neurologicdisease;

rheumatologicaldisease;

obesity;chronicpain;

braininjury;heart

diseases

(congenital);

diabetes,type1;

gastroenterology

Notspecified

5min

Difficult

FS-P

Severityof

fatigue;

impact

offatigue

Cognitive

and

physical

weariness;effort

andassistance

needed

todo

usualactivities;

needingrestand

feelingangry;

avoiding

social

activities

7–18

7–18

(2)

24hor

1wk

Intensity

(14,17,or18

items;14–17

or18–90);

total(18;17–85

or0–68)

5-pointLikert

Hematology/oncology

Notspecified

Notspecified

Easy

PedsQL

3.0CP

Module

(parent)

Notspecified

Notspecified

2–18

2–18

(1)

1mo

Totalfatigue

(4;0–100)

assubdom

aintotalofscale

with

7domains

5-pointLikert

Neurom

otor

Notspecified

30min

including

consent

Dif ficult

PROM

IS(parent)

Severityof

fatigue;

impact

offatigue

Tiredness;lack

ofenergy;impact

onactivities

5–18

1wk

Lack

ofenergy

(11);

tiredness

(23);total

fatigue

(34)

(meanof

50,

SDof

10)

5-pointLikert

or4-point

Likert

(physical

health)

Otherchronichealth

Computer

literacy;

computer

screen;

mouse

access

12–22

min

(PROMIS

I);30–40

min

(PROMIS

II)all

domains

Difficult

EOSQ

Severityof

fatigue

Lack

ofenergy

1–12

4wk

Totalfatigue

(2;n

otspecified)subdom

ainof

scalewith

13domains

5-pointLikert

Musculoskeletal

Notspecified

Notspecified

Difficult

FACIT-F

(modified

parent)

Severityof

fatigue;

impact

offatigue

Notspecified

2–18

1wk

Totalfatigue

(13;0–52)as

subdom

aintotalofscale

with

5domains

5-pointLikert

Hematology/oncology

Notspecified

Notspecified

Easy

PedsQL

ESRD

(parent)

Severityof

fatigue;

impact

offatigue

Notspecified

2–18

1mo

Generalfatigue

(4;0–100)

5-pointLikert

Renaldisease

Notspecified

Notspecified

Difficult

FS-S

Severityof

fatigue;

impact

offatigue

Notspecified

7–18

7–18

(1)

24hor

1wk

Fatigue

intensity

(9,10or

18;9–36)

4-pointLikert

Hematology/oncology

Notspecified

Notspecified

Easy

aBasedon

earlierworkby

Elbers

etal,37

thetheoretical

constructof

fatigue

was

categorizedas

impact

offatigue

ondaily

life,fatigue

severity,orfactorsinfluencingfatigue.


unique in using staff-ratedinstruments (Fig 3).

Dimensions of Fatigue Assessed

Most instruments assessed impact offatigue on daily life and fatigueseverity (14; 70%). Few (4; 20%)assessed fatigue severity symptomsonly, and no instruments assessedimpact of fatigue on daily life inisolation. Although item contentrelated to these dimensions, scoringrarely reflected the dimensionality ofthe constructs assessed, and items formost instruments (14; 70%) weresummed into a total fatigue score.A minority (6; 30%) consisted ofscores for multiple fatigue dimensions:5 child-report measures summeditems into 2 to 4 dimensions, and1 parent report measure summed itemsinto fatigue dimensions (3). There wasinsufficient information on itemcontent for 2 instruments (10%).

Developmental Framework

Overall, participant age was skewedtoward children 12 years and older(Fig 4). Healthy control participantswere included in 23 of 81 studies forwhich age range could be extracted(29%). Self-report of fatigue ischallenging in younger children, andonly 4 (33%) child report instrumentswere identified in use between 5 and7 years of age. Greater administrationburden was commonly noted becauseitems need to be read aloud tochildren or administrators need to beavailable to note children’s verbalresponses. In contrast, 4 (57%) parentreport instruments were identified toassess fatigue symptoms in children asyoung as 2 years of age.

Synthesis of Results

Psychometric Properties

A synthesis summary of theinvestigated methodological

properties of measures andmethodological quality of theincluded studies is provided inTable 6. Full details of the resultantCOSMIN ratings for each study (permeasurement property andquestionnaire) are provided inSupplemental Tables 7–26. Thequality of evidence available acrossquestionnaires varied bymeasurement properties as shown,and no single instrumentdemonstrated positive findings acrossall properties. Content validity wasrated for all instruments and revealedthe most promising evidence.Although assessed in severalinstruments, poor results were foundfor internal consistency. This wasprimarily because studies did not testfor unidimensionality (factoranalysis), or because despitecompleting factor analysis andevidence of more than 1 factor, itemswere summarized into a single score.Cultural validity was not considered,and COSMIN items were used to ratethe quality of the translation acrossall studies with cultural validity data.Few studies revealed informationabout measurement error, reliability,or responsiveness. There was limitedevidence of the floor and ceilingeffects across studies, with ,5% ofincluded studies providinginformation.

There was robust evidence for themeasurement properties of thePedsQL MFS and the FS-C/FS-P. ThePedsQL MFS (child and parentversions) was the only includedinstrument to have strong evidence ofreliability, and the only instrument tohave strong evidence acrossa number of dimensions ofassessment. There were positiveratings for reliability, content validity,and hypothesis testing for the PedsQLMFS. Despite the overall rating forreliability, internal consistencycoefficient (ICC) values varied acrossdifferent populations assessed fromlow to high (eg, 0.27–0.93 for totalfatigue).23,125 Additionally, variabilitywith age has been noted.64 There was

limited evidence for the structuralvalidity of the child version fittinga bifactor model (3 subscales: generalfatigue, sleep-rest fatigue, andcognitive fatigue and a total fatiguescore),122 lending new support toprevious research finding good toexcellent internal consistencystatistics (Cronbach a values between0.70 and 0.95). Strong evidence forhypothesis testing validity for thePedsQL MFS was evident, althoughinternal consistency andmeasurement error of the PedsQLMFS require further research. Thedata for these measurementproperties were gathered in childrenwith numerous health conditions,noted in Tables 3 and 4.

The FS-C, FS-A, and FS-P (standardand 24-hour formats) demonstratedrobust measurement properties,notably content validity. There wasmoderate evidence for the structuralvalidity of the child and parent forms,but limited evidence for thestructural validity of the adolescentform. Internal consistency andreliability data were mixed; althoughinternal consistency statistics weregood (0.70 or greater),79–81,107,113

these statistics were calculated basedon the total fatigue scores, despitefindings of a 3-factor model for theFS-C and a 4-factor model for the FS-A.79,80 One study revealed adequatereliability statistics for the FS-A,presenting pooled data from a seriesof studies, and variable agreementbetween parent and child were foundacross included studies (ICC values0.13–0.65).79 The Fatigue Scale-Staff(FS-S) demonstrated moderateevidence for internal consistency andcontent validity and limited evidencefor structural validity.38,68,75,79 Theassessed evidence for FS-C, FS-A, andFS-P was gathered exclusively instudies of children with hematology/oncology diagnoses, limitinggeneralizability of findings to otherhealth conditions.

The Patient-Reported OutcomesMeasurement Information System

TABLE 5 Respondent Types Within Studies

Respondent No. (%) of Studies

Child only 36 (40)Child and parent 33 (37)Parent only 15 (17)


http://pediatrics.aappublications.org/lookup/suppl/doi:10.1542/peds.2014-2440/-/DCSupplemental

http://pediatrics.aappublications.org/lookup/suppl/doi:10.1542/peds.2014-2440/-/DCSupplemental

(PROMIS)77,88,89,114,123 is a newlydeveloped outcome measure (fatiguesubscale). Among identified studies,those that used PROMIS were theonly ones to employ item responsetheory for statistical analysis. Morepublished information was availablefor the child version, with positiveratings evident for content validityand hypothesis testing. However,there were also some positive

findings for the content and validityof the PROMIS parent report. Overall,properties were given largelyindeterminate ratings because themeasurement properties have not yetbeen evaluated. Evidence wasgathered in study populations ofchildren with mixed healthconditions.

There was limited evidence forhypothesis testing validity of the

Checklist Individual Strength-20 (CIS-20)7,17,101,102,117,119–121 in childrenwith neurologic disorders and chronicfatigue syndrome. Overall findings forthe scale were equivocal.

Some disease-specific instrumentsdemonstrated positive ratings forcontent validity in these specificpopulations (eg, the PedsQL 3.0Cerebral Palsy Module [PedsQL 3.0CP Module]11,12 and the Early Onset

FIGURE 4Age range representation for clinical and healthy participants in included studies (proportion of studies, %). Mean age data for participants set out for81 studies; no mean age participant data reported for 8 studies. Mean age data for controls set out for 26 studies; no mean age control data reported for63 studies.

TABLE 6 Levels of Evidence for the Overall Quality of the Measurement Property per Instrument

Instrument Namea InternalConsistency

Reliability MeasurementError

ContentValidity

StructuralValidity

HypothesisTesting

Cross CulturalValidity

Responsiveness

PedsQL MFS (parent) ? +++ ? +++ ? +++ + ?PedsQL MFS (child) ? +++ ? +++ + +++ + ?FS-C or FS-C 24 h 2 ? ? +++ ++ + + ?FS-A or FS-A 24 h ? 2 ? +++ + + ? ?FS-P or FS-P 24 h ? ? ? +++ ++ + + ?FS-S ++ ? ? ++ + ? ? ?Chalder Fatigue Scale ? ? ? ? ? 2 ? ?CIS-20 ? ? ? ? ? + ? ?PROMIS (child) ? + ? +++ ? ++ ? ?PROMIS (parent) ? ? ? ++ ? ? ? ?FSS ? ? ? ? ? 2 ? ?PedsQL 3.0 CP Module (parent) ? 2 ? ++ ? +/2 ? ?PedsQL 3.0 CP Module (child) ? ? ? + ? + ? ?VAS ? ? ? ? ? ? ? ?POMS ? ? ? ? ? ? ? ?FACIT-F (modified parent) + ? ? + ? ? ? ?Q-sort ? ? ? + ? ? ? ?PedsQL ESRD (parent) ? ? ? + ? ? ? ?PedsQL ERSD (child) ? ? ? + ? ? ? ?EOSQ (parent) ? ? ? ++ ? ? ? ?a Strong +++ or222, Consistent findings in multiple studies of good methodological quality or in 1 study of excellent methodological quality; Moderate ++ or22, Consistent findingsin multiple studies of fair methodological quality or in 1 study of good methodological quality; Limited + or 2, 1 study of fair methodological quality; Conflicting +/2, Conflicting findings;Unknown ?, Only studies of poor methodological quality.


Scoliosis Questionnaire [EOSQ]),62,127

although lacked other evidence forthe quality of their measurementproperties. However, the majority ofincluded instruments demonstratedlow ratings overall. There was limitedevidence for the content validity ofthe Pediatric Functional Assessmentof Chronic Illness Therapy-Fatigue(FACIT-F)33,69,92 and the Q-sort92

instruments for use in hematology/oncology populations, despite nopublished evidence for other assessedmeasurement properties. There wasno positive evidence formeasurement properties of theChalder FatigueScale,58,59,63,90,91,96,106 or the FatigueSeverity Scale (FSS),4,6,73,87 used inchildren with chronic fatiguesyndrome. Often these studies did notreport on measurement properties.There was also no published evidencefor the measurement properties ofthe Profile of Mood States (POMS;hematology/oncology andneurology),104,109 the PedsQL EndStage Renal Disease Module (PedsQLERSD) parent or child (renaldisease),62,71,105,127 nor the includedVASs (hematology/oncology andmusculoskeletal illnesses).66,112

Risk of Bias

Risk of bias was identified acrossstudies for several instruments, dueto reanalysis of participant dataacross studies, including PedsQLMFS,23–25,36,72,93,125 the FatigueScales (child, parent, andadolescent),81,82 and the CIS-20.119,120 Repeated analysis of groupdata possibly skews findings andlimits the generalizability of findingsto other populations. Further,although declared, a primaryresearcher holds the copyright for thePedsQL including PedsQL MFS,23,25,27

PedsQL 3.0 CP Module,11,12 andthe PedsQL ESRD Module.71

DISCUSSION

This study is the first tosystematically review and criticallyappraise fatigue instruments across

the range of childhood chronic healthconditions and ages. Twentyinstruments (12 child reports, 7parent reports, and 1 staff report)were described with reference topatient characteristics (age anddiagnosis), dimensionality of fatigueassessed, and child development. Thisreview provides a thorough andunique review of childhood fatigueinstruments across conditions, usingstandardized published measures toassess the measurement properties ofthese instruments and to assess thequality of the evidence for theseproperties.

In applying COSMIN criteria andassessing the quality of the study,evidence for the measurementproperties of the 20 instruments canbe compared. This review did notidentify any child or parent reportinstrument that met all of the criteria.This appraisal highlights key issues inthe assessment of fatigue inchildhood health conditions. It iswidely acknowledged that asa subjective symptom, fatigue shouldbe assessed by using self report,although developmentalconsiderations necessarily restrict thefeasibility of this in younger children.However, it is important to recognizethe noted lack of robust evidence forreliability. Where ICC values wererecorded, they were often low,indicating lack of agreement betweenchild and parent report onassessment instruments, meaningchild and parent report instrumentscannot be directly compared. It isrecommended that parent and childperspectives be carefully consideredwhen interpreting findings relating tofatigue symptoms in children.

Over 14 health conditions wereidentified as experiencing symptomsof fatigue; however, evidence forinstruments varied across theseconditions. Findings from this reviewsuggest that evidence is limited toinstruments that assess fatigue inchildren with hematology/oncology,and other health conditions.

Specifically, there was support forthe measurement properties of thePedsQL MFS, and limited evidencefor the PROMIS instrument inchildren with a range of chronichealth conditions, and for the FS-C,FS-P, and the FS-A in children withcancer. Evidence for instrumentproperties in children with chronicfatigue syndrome was lacking,despite the high proportion ofstudies with this population. Further,although literature suggests the lowthreshold of fatigue symptomsdistinguishes this clinicalcondition,133 no reviewedinstruments assessed this. Practicingclinicians should be aware thatfatigue can arise from many causesand conditions, and awareness of theunderlying disorder and relevantclinical features is critical toselecting the appropriate validassessment tool.

Whether fatigue should beconceptualized as unidimensional ormultidimensional has been a matterof considerable debate, and evidentlymeasurement of fatigue is a complexissue. Descriptive findings suggestthat this issue is poorly managedby currently available fatigueinstruments. Notably, studies failedto confirm the number of fatiguedimensions (by factor analysis orother methods), or assessment offatigue was conducted by usinginstruments in which the number ofscales did not correspond to thenumber of dimensions identified infactor analysis. For example, of thequestionnaires with the most robustpsychometric properties and studyquality, scores are summed to 3dimensions for the PedsQL MFS, anda total score for the fatigue scales(FS-C, FS-A, and FS-P). However, thereis evidence from factor analysis thatboth instruments are explained by3 to 4 factors.79,80,122 Studies beyondthe scope of this review furthersuggest multiple factors in fatigueassessment instruments, with recentevidence confirming a 3-factorstructure of the PedsQL MFS,134 and


a 2-factor structure of the PROMISscale.2 The field of research is yet todevelop a coherent way of relatingthis emerging evidence regardingfatigue dimensionality withquantification of symptoms. Inclinical practice, it is important torecognize the complexity of fatigue,and to move beyond simplisticassessment of the symptom toconsider a multidimensionalapproach to assessment. Cliniciansshould be aware of the differentinformation provided by the range ofassessment instruments.

This review considered child-specificissues relating to fatigue assessment.However, the findings for reviewedfatigue instruments cannot begeneralized across age rangesbecause participants were mainlyolder than 12 years of age, and gapsin the quality of evidence wereidentified at key developmentalstages. It is important to identify theages in which studies have beenperformed because measurementproperties may vary in different ageranges. Few instruments arevalidated for use in younger children,with robust evidence provided onlyby the PedsQL MFS (age range, 2–18years), and limited evidenceprovided by the PedsQL 3.0 CPModule and the EOSQ. There was

limited reference to healthy controlparticipants in included studies. It iscrucial to the determination ofexcessive fatigue that normaldevelopmental differences are takeninto account because fatigue isa symptom that occurs in healthypopulations, and revealsdevelopmental variation.85,128,135

Further studies are required toexamine fatigue expression acrossage groups, including notablyyounger children, and makereference to normal development, tounderstand how age affects theexperience of fatigue.

LIMITATIONS

Limitations of this review includea focus on children 18 years oryounger with different healthconditions. Evidence ofmeasurement properties in use inother populations was notconsidered. For example, despiteseveral of these measures beingdescribed in older or healthychildhood populations, the inclusionof these findings was beyond thescope of this review. Care should betaken when considering thegeneralizability of the findings toother patient groups. Further, thereview was restricted to subjectivemeasures of fatigue and did not

include studies that have attemptedto measure fatigue objectively.

CONCLUSIONS

There are a large number ofinstruments available to measurefatigue across a range of childhoodhealth conditions, yet there is limitedevidence for their utility. There wasbest evidence for the use of thePedsQL MFS (parent and child report)in a range of pediatric chronic healthconditions across childhood, and theFS-C, FS-A, and FS-P in children withcancer of school age. Use of otherinstruments should be limited toresearch into the validity of theinstrument, not patient outcomes.Further data are required to supportthe measurement properties ofinstruments used to assess fatigue inchildren with chronic fatiguesyndrome, to identify appropriatetools to assess fatigue in childrenyounger than school age, and toclarify the nature of agreementbetween child and proxy reportacross developmental stages.

ACKNOWLEDGMENT

We thank Poh Chua, DeputyLibrarian, RCH, Melbourne, for herhelp in use of the EBSCOhost platformand search terms.

FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.

FUNDING: Supported in part by a Moving Ahead Seed grant, a National Health and Medical Research Council Postgraduate Scholarship (Dr Crichton, awarded 2014),

Canberra, Australia, a Neurosciences Victoria Brain and Mind Scholarship, Melbourne, Australia (Dr Crichton, awarded 2012), and the Victorian Government

Operations Infrastructure Scheme.

POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.

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