Frailty vs. Disability Distinctions in People WithIntellectual Disabilitiesjppi_247 49..58Barbara Brehmer and Germain WeberUniversity of Vienna, Vienna, Austria
Abstract While the extent of research on age-related frailty among adults in general has increased substantially during the past twodecades, there has been a void in equivalent research with respect to adults with intellectual disabilities (ID). While frailty anddisability have been seen as different, but overlapping, concepts in the field of ID, the concept of frailty is missing an empiricalfoundation. The authors define the concept of frailty as applied to adults with ID, using indicators based on Frieds definition (i.e.,a physiological state of increased vulnerability to stressors that follows from decreased physiological reserves and dysregulation of aphysiological system) and Rockwoods frailty criteria (multisystemic instability, change over time, allowance for heterogeneity,association with aging, and association with an increased risk of adverse outcomes). They developed a frailty questionnaire and testedtheir concept of frailty against the concept of disability. To do this, frailty-related data using self-report and third-party interviewswere collected within the Austrian component of the POMONA II Project using a sample of 190 people with ID aged between 18 and76 years. Variables in four frailty domainssocial, cognitive, physical, and mental healthwere assessed and defined as contributingto causal frailty factors. Similarities of the frailty syndrome were analyzed between the general population and the population withID. The results offer evidence of a frailty syndrome affecting older people with ID.
Keywords: aging, falls, frailty, intellectual disabilities, Vienna Frailty Questionnaire for People with ID (VFQ-ID)
Over the past two decades, there has been a substantialincrease of frailty-related publications related to the generalpopulation (Hogan, Mac Knight, & Bergman, 2003; Morley,Perry, &Miller, 2002; Rockwood,Hogan, &Mac Knight, 2000). Inaddition, during this period, the word frailty has been increas-ingly used in the intellectual disability (ID) literature, but its useremained mostly associated with aging issues of family carers(e.g., Barton, 1998). However, though longevity and risks relatedto old age in people with ID have been widely discussed inresearch literature, outcomes of genuine frailty research for the IDpopulation outcomes were not available.
One of the reasons for the dearth of research into frailtyamong adults with ID might be that disability concepts are seenper se as sensitive to specific age-related limitation in functionalcapacities. Furthermore, the emerging concept of frailty is char-acterized by a variety of definitions, with most of them notreflecting the concept of disability in a systematic way, whichprobably has contributed to the lack of a widely accepted defini-tion of frailty (Hogan et al., 2003). In general, frailty definitionsvary between those with a focus on decline in physiologicalsystem functioning (e.g., Cohen, 2000; Hammerman, 1999) and
those promoting an activities of daily living (ADL) or an instru-mental ADL (IADL) foundation (Miles et al., 2001; Nourhashemiet al., 2001). For instance, Campbell and Buchners (1997, p. 315)definition is an essentially physiological conception. They notethat frailty is a condition or syndrome which results from amulti-system reduction in reserve capacity to the extent that anumber of physiological systems are close to, or past, the thresh-old of systematic clinical failure. This definition contrasts withBrown, Renwick, and Raphaels (1995) position, which suggeststhat frailty occurs when there is diminished ability to carry outimportant practical and social activities of daily life. However,there is agreement among researchers that frail older adults aremore vulnerable, have a higher risk for a range of adverse healthoutcomes (e.g., falls and mortality), and increasingly use healthservices and similar resources more often.
A popular definition of frailty has been put forward by Fried(1994). She describes frailty as a physiological state of increasedvulnerability to stressors that follows from decreased physiologi-cal reserves and dysregulation of a physiological system. Shefocuses on the description of the clinical phenotype of frailty,with loss of endurance and body weight; decreased balance,mobility, and cognitive functioning; wasting muscle mass andstrength; slowed performance; and relative inactivity consideredas core symptoms. The assessment strategy refers to self-reporteddata as well as performance tests of five elements: weakness,weight loss, slow walking speed, fatigue, and low levels of activity.
Within the dynamic model of frailty as suggested by Rock-wood, Fox, Stolee, Robertson, and Beattie (1994), frailty is viewed
Received July 31, 2009; accepted October 18, 2009Correspondence: Barbara Brehmer, Institute of Clinical, Biological andDifferential Psychology, University of Vienna, Liebigg 5, Vienna, 1010Austria. Tel: +43 14277 47896; E-mail: email@example.com
Journal of Policy and Practice in Intellectual DisabilitiesVolume 7 Number 1 pp 4958 March 2010
2010 International Association for the Scientific Study of Intellectual Disabilities and Wiley Periodicals, Inc.
as if it were a balance beam, in which the balance between assetsand deficits determines whether a person can maintain indepen-dence in the community. Clearly, this perception of frailty is,unlike that of Frieds, not solely focused on physiological symp-toms but one that views frailty as a dynamic, complex interactionof biological, psychological, cognitive, and social factors. There isno disagreement with the clinical phenotype of Fried. Bothresearchers agree that frailty is associated with aging. Frailty,according to both workers, is characterized by fluctuations ofhealth status, which result successively in negative health out-comes. In addition, Rockwood et al. (2000) suggested five criteriato be included in a frailty definition: multisystemic instability,change over time, allowance for heterogeneity within a popula-tion, association with aging, and association with an increasedrisk of adverse outcomes.
Historically, frailty has been equated with disability(Fried, Ferrucci, Darer, Williamson, & Anderson, 2004; Rock-wood et al., 2000), but more recently, researchers haveattempted to differentiate these two concepts, leading to con-ceptual disjunctions. Fried, Tangen, and Walston (2001) statedthat disability often refers to the inability to independently carryout instrumental and basic ADL. They concluded frailty anddisability to be distinct but overlapping concepts. So, not allolder persons with disabilities are frail and not all frail eldershave disabilities. Disability, though, can be an adverse outcomeof frailty, and if frailty is the pathway to disability for an indi-vidual, he or she does not cease to be frail with the onset ofdisability. This notion is supported by results of a poll among 62geriatricians (Fried et al., 2004). However, Frieds reference todisability is limited to physical disabilities, and until now, therehas been no evidence for these findings with respect to peoplewith ID.
The aim of the present study was to analyze frailty, definedas a multisystemic instability, in a population of people with ID.The Vienna Frailty Questionnaire for People with ID (VFQ-ID)based on Rockwoods criteria and Frieds description of frailty forthe general population was developed by the authors and used tocollect data for this study.
This study was included in the protocol of the EuropeanUnions POMONA II projects Health Indicators for People withIntellectual Disabilities (Brehmer, 2008). The aim of POMONAII was to test a set of 18 health indicators for adults with ID in 14European countries using a common POMONA questionnaire.The health indicators were developed in the previous POMONAI study and were structured in the following four domains (VanSchrojenstein Lantman-deValk, Linehan, Kerr, & Noonan-Walsh,2007): health status (e.g., epilepsy, mobility, sensory and bodymass index (BMI)); health systems (e.g., health check and healthpromotion); health determinants (e.g., physical activity and chal-lenging behavior); and health demographics (e.g., daily occupa-tion and income). As the participant members of the POMONAII study were free to extend the protocol with additional researchareas, the authors undertook to examine frailty using a speciallydesigned questionnaire.
Sampling and Participants
The sampling procedure of the POMONA II project aimed atcreating equivalent group sizes with respect to intensity ofsupport (disability level), age, sex, and accommodation setting(with an overall total of 80 subjects) in order to test reliability andfeasibility of the POMONA II protocol for various subgroups ofpeople with ID. However, the study of frailty required an amend-ment of the Austrian sampling procedure in order to fulfill majorcriteria for representativeness (Brehmer, Zeilinger, & Weber,2008). Thus, a total number of 204 people with ID from eighthealth areas in Austria (among them are Vienna,Waldviertel andWeinviertel) participated in the study. Usable data were receivedfrom 190 adults. The Austrian POMONA sample was consideredrepresentative of the Austrian ID population with respect to theseparameters (age, living and geographic setting, and capacity).Their age range was 1876 (the floor was set by the POMONA IIproject) with a mean age of 41 years. Some 60% (n = 114) residedin an institution or a group home. The balance (40%; n = 76)either lived alone or with their family.With respect to geographiclocation, 67.4% resided in an urbanized area and 32.6% resided ina rural area. With respect to intensity of support (i.e., level ofdisability), 77.9% were able to speak for themselves during theinterview, showed characteristics for independent living, and hadlow needs of support; 22.1% were nonverbal, showed prominentcharacteristics for dependency, and had high needs of support(level of disability was not objectively measured in the POMONAII project and institutional records showed to be of little reliabilityin this regard). There was no preselection for sex, as an equaldistribution was expected. Our sample resulted in 98 men and 92women (Table 1).
Procedures and Data Collection
Once the POMONA II protocol was accepted by the Univer-sity of Viennas Ethics Board, queries were sent to 25 Austrianservice and support provider organizations located in Vienna andLower Austria asking whether they would help the authors byenabling some of their clientele to participate in the study. Fifteenof these service provider organizations agreed to be part of theproject and to help identify study subjects. The participating pro-viders were asked to nominate adult clients who met our studycriteria, inform them about the study, and then help the authorssolicit their voluntary participation. Once potential subjects wereidentified, they were queried by the staff at the provider organi-zations as to their willingness to participate in the study and, ifagreeable, introduced to the studys interviewers. The interview-ers were eight graduate and postgraduate psychology students atthe University of Vienna, most of them with prior front line staffexperience with ID provider organizations. They were speciallytrained for the POMONA II interviews by the authors, with spe-cific attention given to procedures and competences for establish-ing informed consent and in extracting data during interviews.
In a first face-to-face meeting, interviewers explained thebackground, aims, and contents of the project to the people withID, their support persons, and their legal guardians. The inter-viewers also informed everyone by means of an easy-to-readinformational letter about anonymity and participants rights
Journal of Policy and Practice in Intellectual Disabilities Volume 7 Number 1 March 2010
B. Brehmer and G. Weber Frailty and Intellectual Disabilities
(i.e., they could withdraw at any time of the interview, have abreak during the interviews, refuse to answer, and be supportedby a personal assistant). In addition, the persons with ID had theright to define the time and place for the interviews. As thePOMONA II protocol contained self-reported and third-partydata (i.e., information from the primary carer or a familymember), participants and their legal guardians were also askedto give consent for this procedure. Health information aboutnonverbal participants was obtained only through third parties.The consent procedural part of the interview ended with anagreement on the date and time for the formal study interview.The average formal study interview duration was 1 h for personswith ID, with an additional hour allocated for collecting datafrom third parties. Data were collected by the eight interviewersover a year (from November 2006 to November 2007).
Assessment and Instruments
The development of the VFQ-ID, a frailty measure, was basedon in-depth analysis of approximately 200 published articles onfrailty, some mentioning Rockwoods criteria or using Friedsdefinition. These two frameworks were assumed to be mostusable for studying the frailty syndrome in the population withID. The analysis suggested the development of a new assessmenttool for several reasons: (1) there was significant heterogeneity offrailty measurement strategies because of a lack of acceptedoperational definitions; (2) no instrument fulfilled Rockwoodscriteria; (3) instruments that could assess multisystemic impair-mentswere lacking; (4) most instruments included only physicalsymptoms, and only rarely were cognitive or psychological healthoutcomes considered (only one measurement (Schuurmans,Steverink, & Lindenberg, 2004) included social factors); and (5)all instruments were constructed to measure the status quo andwere used in longitudinal studies to evaluate negative healthchanges. Given this, it was decided to construct a unique frailtyquestionnaire that would be useful in assessing change. Further,as the instrument needed to be designed to gather information onfrailty as perceived by people with ID, or as viewed by third parties,
objective performance measurements were not included. Thisallowed screening for frailty by nonprofessionals. In addition, thenew questionnaire needed to fulfill two extra requirements: itneeded to include criteria that could be operationalized and itneeded to include variables that were reported in prior research todescribe and assess frailty.
Operationalizing frailty Consistent with the literature analysis,three of the five Rockwood criteria (i.e., multisystemic instability,change over time, and association with an increased risk ofadverse outcomes) can be detected as central when assessingfrailty. Frailty has an unstable influence on four different systems:physical (e.g., washing and eating), psychological (e.g., me...