Measuring the psychosocial health of adolescent and young adult (AYA) cancer survivors: a critical review

Cancer is the leading disease-related cause of mortality among adolescents and young adults (AYAs) resulting in approximately 134,000 deaths worldwide, each year [1]. AYAs have been broadly defined as young people between the ages of 15 and 30 years [2‐4]. Advances in treatment mean that between 73-82% of AYA diagnosed with cancer will now survive up to five years post-diagnosis [5‐8]. Increasing survival rates mean that a greater number of AYAs are living longer with the psychosocial sequelae of their cancer diagnosis and its treatment [7‐10]. AYAs not only experience the wide range of physical, psychological, social and spiritual concerns of cancer survivors of all ages, but often have additional and unique needs due to their cancer occurring during a crucial stage of their personal and social development [11‐13].

Cancer survivorship has been defined as beginning from the time of cancer diagnosis and includes people at various stages of the disease trajectory [14]. Although grouped due to their unique developmental phase, AYA cancer survivors represent a variety of socio-demographic backgrounds and cancer types. Some AYA survivors include students who live with their families, while others are employed and live independently [15, 16]. The majority have a history of lymphoma, leukaemia, invasive skin, genital, endocrine, brain or bone cancer [4, 6‐8].

The acute psychosocial impact of cancer and its treatment may be substantial. Some AYAs experience physical side-effects such as pain, vomiting, and nausea [17, 18]. These physical symptoms can lead to high levels of distress in young people, and can limit their ability to engage in normal activities such as attending school or work [15]. Participation in social events is often restricted, and can mean that normal adolescent rites of passage, such as the formation of identity and independence are inadequately achieved [12, 19, 20]. This lack of social interaction with peers can lead to feelings of isolation and loneliness [11, 12]. Side-effects of treatment such as weight loss, hair loss or impaired physical development can impact on perceived body image and can contribute to loss of self-confidence [12, 19, 21]. Feelings of hopelessness or anxiety have also been reported [2, 22]. A young person's cancer diagnosis can also lead to changes in family dynamics and impact on their relationships with parents, siblings, and significant others [12, 13, 19].

Although some acute psychosocial consequences cease once treatment is completed, others can have a long-term impact on the psychosocial health of the survivor. Compared with other young people their age, some long-term AYA cancer survivors report poorer health outcomes including higher rates of obesity, anxiety and depression [20, 23, 24]. Some also experience cognitive impairment which can impact on employment and educational attainment [25, 26]. Concerns related to reduced fertility and sexual dysfunction are also prevalent among AYA cancer survivors [27, 28].

The widely accepted World Health Organisation (WHO) definition of health encompasses physical, mental and social aspects of well-being, all of which are inextricably linked and contribute to the global health of the individual [6]. This necessitates the use of multi-dimensional rather than uni-dimensional measures in order to develop a comprehensive assessment of the health of an individual [29]. Multi-dimensional measures of health assess elements of physical, psychological, social, and often spiritual well-being [29]. For cancer patients these generally include measures of quality of life (QoL) and perceived need. QoL measures assess an individual's perception of their current health status compared with their health expectations [29, 30]. In contrast, measures of perceived need identify the needs individuals regard as being unmet and the magnitude of help likely to be required to address them [31, 32]. While there are a number of QoL and unmet needs tools for adult cancer patients and survivors [33, 34], few measures specific to AYA cancer survivors have been identified [35‐39]. Given the unique needs and experiences of this group, psychosocial health measures developed and validated with this population are needed to accurately assess well-being.

Self-report rather than proxy measurement is generally preferred for assessing psychosocial health. Although proxy measurement may allow for the inclusion of patients who are too ill or do not have the necessary literacy skills to participate alone, proxies can tend to base their assessment on their impression of the patient, rather than the actual situation [40, 41]. Proxies are also more inclined to focus on negative or extreme behaviour rather than positive or usual behaviour [41].

As well as being assessed by self-report and covering broad psychosocial domains, measures designed to assess the psychosocial well-being of AYA cancer survivors need to be able to accurately reflect the unique experiences of this population. Such measures should be able to capture, and be sensitive to, changes in psychosocial health across the disease trajectory so that the effectiveness of interventions can be assessed [38]. Measures also need to be psychometrically robust so that the prevalence of needs, and subgroups of young people experiencing high needs, can be accurately identified [38].

The aim of this review is to critically examine the psychometric properties of multi-dimensional, self report measures developed to assess the psychosocial health of AYA cancer survivors.

Medline, PsychINFO, EMBASE and CINAHL databases were searched to identify publications which described the development of measures for assessing psychosocial outcomes in AYA cancer survivors. These databases were chosen as they all provide extensive coverage of journals in the field of cancer research.

The database search was performed using the following combinations of keywords: [neoplasm or cancer or oncol*] and [adoles* or teenager or young adult or youth] and [perceived need* or unmet need* or quality of life or psychosocial or distress] and [develop* or questionnaire or survey or measure or scale] and [psychometric or reliability or validity or acceptability]. Results of the search were limited to the English language and covered the last ten years from 1998 to 2008. This timeframe was selected as a preliminary search of Medline for all AYA related psychosocial research without a year limitation revealed that there had been minimal (< 17%) research output in field prior to 1998 (Figure 1), with only one publication before 1988 identified (one publication in 1976). Appraisal of these 23 publications revealed that no additional measures met the inclusion criteria (outlined below) prior to 1998.

Duplicate publications, and publications which did not specifically describe the development, psychometric properties, or acceptability of a measure, were excluded. Full text articles of the remaining publications were obtained and reviewed to identify relevant measures.

While AYAs are commonly defined as 15-30 year olds, definitions in the literature vary [2‐4]. Therefore, an inclusive approach was employed whereby scales developed for use with young people less than 15 years but with an upper age limit between 15 and 30 years were included (eg 12-20 years). Similarly, scales developed for use with populations older than 15 years but less than 30 years were included (eg 16-28 years).

Measures which met all of the following criteria were included in the study for coding: 1) quantitative; 2) developed or validated in English; 3) multi-dimensional and measured at least the following three psychosocial domains: physical, psychological, and social; 4) cancer specific; 5) assessed the well-being of patients or survivors; 6) developed specifically for AYA or included participants aged between15-30 years in their sample; and 7) completed by self-report.

After identifying measures which met all of the inclusion criteria, a second search of all databases by 'measure name' was performed to ensure that all publications relating to each identified measure were obtained.

One reviewer used the inclusion and exclusion criteria to identify measures for inclusion in the review. A second reviewer cross-checked 15% of the measures, to confirm their inclusion and exclusion status. The psychometric criteria of all included measures were reviewed by the first author and checked by the second.

The initial search of the Medline, PsychINFO, EMBASE and CINAHL databases identified a total of 552 publications related to assessing psychosocial outcomes in AYA cancer survivors, with 436 papers having been published in the last ten years (1998-2008). Of these 436 publications, 91 were duplicates and 146 did not describe the development of a measure. The remaining 199 publications described the development of 204 measures.

197 measures did not meet the inclusion criteria (Figure 2), leaving seven measures to be included in the psychometric review. These included the: 1) Adolescent Quality of Life Instrument (AQoL)[35, 36]; 2) Minneapolis-Manchester Quality of Life Instrument (MMQL) - Adolescent Form [52‐54]; 3) Pediatric Quality of Life Inventory (PedsQL) 3.0 Cancer Module Child and Adolescent (C&A) Forms [55‐58]; 4) Quality of Life - Cancer Survivors (QOL-CS) validation in childhood cancer survivors [16]; 5) Pediatric Cancer Quality of Life Inventory - 32 Short Form (PCQL-32) [59‐61]; 6) Pediatric Cancer Quality of Life Inventory (PCQL) Modular Approach [62]; and 7) Perceived Illness Experience Scale (PIE)[63, 64].

Six measures were developed in the United States, one was developed in the United Kingdom [63, 64]. A description of each measure's domains and number of items is presented in Table 2.

Overall, reporting of the sample accrual method and the sociodemographic and clinical characteristics of participants for each measure was comprehensive (Table 3). Of the seven measures, three did not report a response rate, one did not describe the inclusion and exclusion criteria, and one measure did not report the proportion of male and female participants or cancer type.

All measures were developed using samples recruited through hospitals or medical centres. Sample sizes ranged from 41-291 participants, and age of participants ranged from 5-28 years (mean range of 10.9-21.8 years). The proportion of males and females was reasonably equally distributed. For the majority of studies, the greatest proportion of young people had been diagnosed with Leukaemia. Cancer treatment stage ranged from newly on treatment to 3-27 years post-diagnosis.

An overall summary of the psychometric properties reported for each measure can be seen in Table 4.

Only two measures reported floor and ceiling effects (Table 7). None of the measures reported their ability to detect clinically important change over time.

Table 7 also shows that the acceptability of the measures was poorly described with only four measures reporting missing items, and only three measures reporting their reading level. The reading levels that were reported however were appropriate for the population group. Feasibility, the time needed to administer, complete, and score the measure, was not reported for any of the measures.

Two measures, the MMQL Adolescent Form and PedsQL 3.0 Cancer Module (C&A), have been adapted for cultures other than the United States. For the culturally adapted measures, similar reliability and validity to the original measure was reported. The reliability of MMQL Adolescent Form in an online format has also been verified.

The literature search for this review was conducted using four online publication databases, and the grey literature was not included. Therefore, it is possible that some relevant measures were missed. However, it is likely that measures identified in this review are likely to be of the best quality as they have been published in peer-reviewed, indexed journals. The step of conducting a second search by measure name would have also minimised the chance that publications relating to relevant measures were overlooked.

The definition of AYA cancer survivors used in this review was young people between the ages of 15 and 30 years. However as a group, the AYA population is not defined well in literature, and ranges from 12 up to 40 years [2‐4]. To overcome this discrepancy, any measures developed for an age cohort which overlapped the 15 to 30 year old age bracket were included. This may mean that some of the results reported in this review reflect measure performance with individuals outside the AYA definition used for this review.