Pediatr Blood Cancer 2011;56:439–443

Prospective Evaluation of Legal Difficulties and Quality of Lifein Adult Survivors of Childhood Cancer

Robert Olson, BSc, MD, FRCPC,1,2* Gabe Hung, BSc,3 Mary Anne Bobinski, BA, JD, LLM, PhD,3

and Karen Goddard, FRCP(UK), FRCPC4

INTRODUCTION

Over the past several decades, therapeutic advances in the

treatment of pediatric malignancies have led to significant improve-

ments in overall patient survival. Currently, at least 80% of all

children with pediatric malignancies will be cured of their disease

[1]. As a result, it is estimated that 1 person in 250 of the adult

population is currently a survivor of childhood cancer [2].

Improvements in cure rates are predominantly attributed to

therapy intensification, including increased chemotherapy dose and

intensity, more aggressive surgery, and the concurrent adminis-

tration of chemotherapy with radiotherapy. However, therapy

intensification increases the risk of long term side effects, which

have the potential to negatively impact survivors’ long term health

and wellbeing [3]. This has been documented extensively in the last

decade [4–9]. While the majority of patients will have mild to

moderate health problems, at least 40% of survivors have late effects

related to their previous cancer and therapy that significantly affect

their health related quality of life (QoL) [10].

Educational and social problems that persist into adulthood are

common and particularly problematic late side effects in adult

survivors of childhood cancer (ASCC) [11]. As a result, survivors

are likely to be at increased risk of legal difficulties (LDs), including

discrimination and limited access to public services, such as

appropriate education, rehabilitation, employment, disability bene-

fits, dental plans, and insurance [12–15]. Because of their increased

risk of side effects, we hypothesize that survivors of CNS tumors are

most at risk of LDs. We also hypothesize that LDs negatively affect

the QoL of ASCC.

PATIENTS AND METHODS

Survey Construction and QoL Measure Selection

A survey was developed at the BC Cancer Agency (BCCA), in

order to collect information on survivors’ legal needs. Content of

the survey was initially developed by the study authors, but subse-

quently modified after patient feedback. Specifically, survivors’

opinions regarding difficulty with the cost of prescriptions were

added after 10 patients had already completed the questionnaire.

Our primary objective was to assess the relationship between

QoL and LDs. Therefore, the Functional Assessment of Cancer

Therapy (FACT) was completed by survey respondent in order to

assess QoL. The FACT is a well validated and commonly used

assessment of QoL in both general, and central nervous system

(CNS) oncology [16–18]. Survivors were asked to complete the

assessment themselves, though permitted to seek assistance from

family, friends, or caregivers.

Patient Selection

One hundred twelve patients were consecutively approached at

the BCCA Late Effects Clinic (LEC) from January 2009 to February

2010. All patients who attended the clinic were considered eligible,

and approached. Patients who attend the clinic are followed because

they are at intermediate or high risk of grades 3 or 4 late effects

as a result of their therapy [19–21]. Only 1 patient declined to

participate.

Statistics

A convenience sample size of 100 subjects was selected for this

study based on the estimated number of patients that would be seen

during the study period. Descriptive statistics were used to present

Background. Adult survivors of childhood cancer (ASCC),especially those of the central nervous system (CNS), have increasedrisks of educational and social difficulties. It is therefore hypothe-sized they are more likely to encounter legal difficulties (LDs), suchas workplace discrimination and disability insurance denials, whichmay negatively affect their quality of life (QoL). Procedure. Wedeveloped a survey to collect information on patients’ legal needs.QoL was assessed using the Functional Assessment of CancerTherapy (FACT). Results. We prospectively approached 112 ASCC,111 (99.1%) of whom completed the survey. The median age ofrespondents was 7 years at diagnosis and 31 years at surveycompletion. CNS tumors were the most common malignancy(32.4%). LDs were common overall (40.7%), though more prevalentin patients with CNS versus non-CNS tumors (58.6% vs. 32.3%;

P¼ 0.023). The most prevalent LD was workplace discrimination(58.3%). On multivariate analysis, CNS tumor was the only variablesignificantly associated with LDs (OR¼ 4.49, P¼ 0.041). Individualswith LDs had lower QoL scores compared to those without LDs(79.96 versus 91.83 on the FACT; P¼0.005). On multivariateanalysis, individuals with LDs had lower QoL scores (14.95 pointslower on the FACT), which is both clinically and statisticallysignificant (P¼0.047). Conclusions. Legal difficulties are commonin adult survivors of childhood cancer, especially those with braintumors. Furthermore, individuals with legal difficulties have worsequality of life. Research is needed to develop effective and accessiblelegal resource programs. Pediatr Blood Cancer 2011;56:439–443. � 2010 Wiley-Liss, Inc.

Key words: brain tumors; legal difficulties; pediatric cancer; quality of life; survivors; workplace discrimination

� 2010 Wiley-Liss, Inc.DOI 10.1002/pbc.22777Published online 9 September 2010 in Wiley Online Library(wileyonlinelibrary.com)

——————1BC Cancer Agency, University of British Columbia, Vancouver, BC,

Canada; 2Department of Epidemiology, Harvard School of Public

Health, Boston, MA; 3University of British Columbia, Vancouver,

British Columbia, Canada; 4BC Cancer Agency, University of British

Columbia, Vancouver, British Columbia, Canada

Conflict of interest: Nothing to declare.

*Correspondence to: Robert Olson, BC Cancer Agency, 600 West

10th Avenue, Vancouver, BC, Canada V5Z 4E6.

E-mail: rolson2@bccancer.bc.ca

Received 22 June 2010; Accepted 13 July 2010

patient characteristics and survey results. Fisher’s exact test

assessed the relationship between categorical respondent variables

and the proportion with LDs. The Mantel–Haenszel common odds

ratio (OR) assessed the magnitude of risk for LD for each categorical

variable. Logistic regression modeling was used to assess the

relationship between LDs and CNS radiotherapy and CNS tumors,

after controlling for other potential confounding variables. Differ-

ences in FACT scores by categorical variables were assessed with

t-tests. Wilcoxon rank sum test assessed the relationship between

potential categorical variables and the number of annual family

physician (FP) visits, since they were non-normally distributed.

Spearman’s correlation coefficient assessed the correlation between

the number of FP visits and QoL scores on the FACT. One way

Analysis of variance (ANOVA) assessed the relationship between

FACT scores and the different categories of FP visits per year.

Multivariate analysis of the relationship between LDs and QoL was

performed using linear regression modeling using propensity score

analysis, because of the small number of outcome events relative to

the number of potential confounding variables [22]. All analyses

were conducted using SPSS Statistics 17.0 software or SAS for

Windows version 9.2.

Research Ethics Approval

The study was approved by the Research Ethics Board at the

BCCA, and was performed in accordance with ethical standards laid

down in the 1964 Declaration of Helsinki. All subjects gave their

informed consent prior to their inclusion in the study. No potential

conflicts of interest exist.

RESULTS

Patient Characteristics

The median age of respondents at diagnosis was 7 years

(Interquartile Range [IQR]¼ 3–11), and 31 (IQR¼ 26–36) at

survey completion. The median interval between diagnosis and

survey completion was 25 years (IQR¼ 19–28). Additional patient

characteristics are presented in Table I.

Survey Response Rate

Of the 112 patients approached, 111 (99.1%) completed the

survey. Despite this excellent response rate, there was considerable

variability in the individual question response rate (range 10.8–

100%; Table II). Of note, 91 (82.0%) of respondents volunteered

whether they had legal difficulties (LDs) or not (Table II). Similarly,

87 (78.4%) of respondents completed the Functional Assessment of

Cancer Therapy (FACT).

Survey Results

Table II presents the survey results, which demonstrates that

LDs are common in adult survivors of pediatric malignancies.

Thirty six of the 37 respondents (97.3%) with LDs provided written

descriptions of the LDs they experienced. The predominant LD

identified from these comments were occupation related (58.3%),

including difficulty finding employment, discrimination at work,

and unfair termination of employment. LDs at school were the next

most common (22.2%), followed by difficulty acquiring life, health,

or disability insurance (16.7%).

Seventeen respondents provided written descriptions of the

legal help they utilized. No consistent resources were identified.

Instead, community organizations, coworkers, employers, tutors,

social workers, lawyers, physicians, friends, and family members

were identified as potential aids to LDs.

Predictors of Legal Difficulties (LDs)

Table III presents the univariate analysis of potential risk factors

for LDs. Notably, respondents with CNS tumors and individuals

who received CNS radiotherapy have a significantly higher

proportion of LDs. This represents an odds ratio (OR) of 2.98

(95% CI¼ 1.20–7.40) for ASCC who had CNS versus non-CNS

tumors. Likewise the OR for ASCC who received CNS RT

compared to those who did not was 3.37 (95% CI¼ 1.12–10.08).

On multivariate analysis, CNS tumor was the only variable signi-

ficantly associated with LDs (Table IV).

Relationship Between Legal Difficulties (LDs)and Quality of Life (QoL)

The overall mean QoL score on the FACT was 87.47 (SD¼16.59). Higher scores represent better QoL [16]. As hypothesized,

individuals with LDs had significantly lower QoL scores on

the FACT than individuals who did not have LDs (Table V). In

addition, females had lower QoL scores than males (Table V). There

were no other significant relationships identified between patient

variables and QoL (Table V). On multivariate analysis, controlling

for age at diagnosis, current age, tumor type, chemotherapy,

Pediatr Blood Cancer DOI 10.1002/pbc

TABLE I. Patient Characteristics

Characteristic

Survivors (n¼ 111)

No. %

Gender

Male 55 49.5

Female 56 50.5

Diagnosis

CNS 36 32.4

Leukemia 32 28.8

Lymphoma 13 11.7

Sarcoma 18 16.2

Wilm’s 7 6.3

Other 5 4.5

Age at diagnosis (years)

<2 14 12.6

2–5 28 25.2

6–10 38 34.2

>10 31 27.9

Current age

<30 61 55.0

�30 50 45.0

Treatment

Chemotherapy 81 73.0

Radiotherapy 102 91.9

CNS radiotherapy 81 73.0

Relapsed malignancy 13 11.7

Secondary tumor 14 12.6

CNS, central nervous system.

440 Olson et al.

radiotherapy, CNS radiotherapy, presence of family physician,

relapse, secondary malignancy, and gender, LDs were associated

with QoL scores 14.95 points lower on the FACT, which is both

clinically and statistically significant (P¼ 0.047).

Visits to Family Physician (FP)

Respondents visited their FP a median of 3 times/year, though

there was significant variability in frequency (0–36 visits/year). The

number of visits per year was inversely related to QoL (r¼�0.252,

P¼ 0.025); in other words, individuals with lower FACT scores

were more likely to see their FP repeatedly. Individuals who saw

their FP <3, 3–10, and >10 times/year had FACT scores of 93.1,

84.8, and 70.3, respectively (P¼ 0.002). In addition, individuals

whose malignancies have relapsed, visit their FP more often than

individuals whose malignancies have not relapsed (median 6 vs.

3 visits/year; P¼ 0.047). Although females saw their FP more often

than male survivors (median 4 vs. 2 times/year), this was not

statistically significant (P¼ 0.209). Likewise, there was no statisti-

cally significant relationship between number of annual FP visits

and LDs (P¼ 0.335), use of chemotherapy (P¼ 0.477), previous

radiotherapy (P¼ 0.901), CNS tumor (P¼ 0.407), CNS RT

(P¼ 0.491) or secondary tumor (P¼ 0.467).

DISCUSSION

Legal Difficulties (LDs) Are Common

Our results indicate that ASCC have a high prevalence of

self perceived LDs, especially those treated for CNS tumors

(Tables II–IV). To the best of our knowledge, this is the first

study to demonstrate this relationship. As hypothesized by

Monaco et al. [12], difficulties are predominantly work or

school related, though also include difficulties obtaining health,

disability, and life insurance. In addition, workplace discrimination

was more common in our study than in previous studies of adults

with cancer, though comparison across studies is problematic,

especially given the different outcome assessments utilized

[15,23,24].

Not only are LDs common, but difficulty finding legal aid

was also frequently reported in our study (Table II). In addition,

the legal aid used by respondents was inconsistent, suggesting

that an easily accessible, common resource would be beneficial.

This is further supported by the fact that 83.0% of respondents

indicated a legal help service for survivors would be useful

(Table II). Furthermore, our results indicate that LDs impose

financial consequences in a significant proportion of respondents

(Table II).

Pediatr Blood Cancer DOI 10.1002/pbc

TABLE II. Selected Survey Results

Question No. of ‘‘yes’’ No. of answered % ‘‘yes’’

Do you have a family doctor? 103 111 92.8

How often do you see your family doctor each year?

0–2 47

101

46.5

3–5 30 29.7

6–10 24 23.8

>10 11 10.9

Have you had any problems with legal issues or discrimination? 37 91 40.7

More than 1 problem? 5 12 41.7

Did you suffer financially as a consequence of this problem? 18 24 75.0

For this problem, was it difficult to find legal help? 18 20 90.0

Did legal help meet your needs? 13 17 76.5

Have you ever had problems with the cost of prescriptions? 24 97a 24.7

Have you ever applied for a disability pension? 29 111 26.1

If yes, was it hard to apply for a disability pension? 9 27 33.3

Would a ‘‘legal help’’ service for survivors be useful? 83 100 83.0

aAdded after 10 subjects.

TABLE III. Relationship Between Patient Variables and LegalDifficulties

Variable

% with Legal Difficulties

If yes If no P-Value

Diagnosis age< 5 44.8 38.7 0.650

Chemotherapy 36.8 54.5 0.212

CNS RT 47.0 20.8 0.030

CNS tumor 58.6 32.3 0.023

Female 44.7 36.4 0.523

Relapse 33.3 41.8 0.755

Second malignancy 54.5 38.8 0.344

CNS, central nervous system; RT, radiotherapy.

TABLE IV. Multivariate Analysis of the Relationship BetweenLegal Difficulties and Patient Variables

Variable

Odds ratio

(95% CI) P-Value

Age at diagnosis (continuous) 0.91 (0.80–1.03) 0.12

Age at accrual (continuous) 1.04 (0.97–1.12) 0.28

Chemotherapy (vs. none) 1.31 (0.31–5.59) 0.72

CNS RT (vs. non-CNS RT) 1.76 (0.48–6.40) 0.39

CNS tumor (vs. non-CNS tumor) 4.49 (1.06–19.04) 0.04

Female (vs. male) 1.64 (0.62–4.39) 0.32

Relapse (vs. none) 0.43 (0.10–1.87) 0.26

Second malignancy (vs. none) 0.76 (0.17–3.35) 0.72

CI, confidence interval; CNS, central nervous system; RT, radiotherapy.

Legal Problems in Pediatric Cancer Survivors 441

Survivors of Central Nervous System (CNS) TumorsHave More Legal Difficulties (LDs)

As hypothesized, LDs were most common in individuals with

CNS tumors and in those who had received cranial radiotherapy

(Table III). Furthermore, after controlling for potential confounding

variables, CNS tumors continued to predict for LDs (Table IV). We

propose that this relationship can be explained by the increased

social and education difficulties experienced by patients with CNS

tumors, as a result of their brain tumors, aggressive multimodality

treatment, and time absent from school [3,4,7,9,25,26].

Our analysis is limited by the number of subjects presenting to

our provincial LEC at the BCCA. Other patient variables listed in

Table III are likely associated with LDs, but our study may have

insufficient statistical power to identify the relationships. Therefore,

we believe that multi-institutional studies in survivors of childhood

cancer are warranted in order to fully explore these potential

relationships.

LDs Are Correlated With Decreased QoL

As hypothesized, individuals with LDs have significantly lower

QoL scores on both univariate and multivariate analysis. This is

likely explained by LDs negatively impacting the daily activities

of respondents, which subsequently decreases their QoL. However,

limitations inherent to cross sectional surveys, prohibits concluding

that LDs are the direct cause of decreased QoL. Most importantly,

without data on LD timing of onset in relation to QoL changes,

the exploration of causality is limited [27].

Generalizability

Given the excellent response rate in our study, we believe that our

results are generalizable to ASCC at intermediate to high risk of

grades 3 or 4 late effects, since these are the patients generally

followed at our provincial LEC [20,21,28,29]. In contrast, we do not

believe these results are generalizable to all ASCC. For example,

patients not followed at our LEC, such as those with low risk

leukemias, treated with less intense chemotherapy alone and no

cranial radiotherapy, are unlikely to have as elevated a risk of LDs.

Furthermore, several questions in our survey had a low response

rate, and therefore are less generalizable, which is why we included

this information in Table II [28,30]. Despite these limitations,

the relationship between CNS tumors and LDs is both highly

statistically and clinically significant.

Future Directions

Our results indicate that LDs are common in survivors of

pediatric brain tumors, which we hypothesize is a result of

neurocognitive deficits in these patients. Therefore, we believe

future research should investigate this potential relationship, and

whether preventing or treating neurocognitive deficits reduces the

risk of LDs. Future research is also needed to investigate if screening

for LDs and offering potential interventions is beneficial. Possible

interventions include the provision of specific counseling and

education services for ASCC in the LEC and the development of a

legal aid service. Given our results, it is likely that improved

identification and management of legal difficulties will improve the

quality of life of survivors of childhood brain tumors.

CONCLUSION

Legal difficulties are common in ASCC, especially those with

CNS tumors. In addition, legal difficulties are correlated with worse

quality of life. Furthermore, lower quality of life is associated

with increased use of resources, including family physician visits.

We believe these results justify the development of a legal resource

centre in British Columbia for adult survivors of pediatric

malignancies. We plan to pursue this centre as a joint collaboration

between the BC Cancer Agency, the BC Children’s Hospital, and the

University of British Columbia’s Faculty of Law. Furthermore, we

believe that research should focus on: (1) confirming our findings,

(2) Studying the relationship between neurocognitive function and

the prevalence of legal difficulties (3) exploring the relationship

between legal difficulties and quality of life, (4) improving access to

legal resources, and (5) improving the quality of legal resources for

ASCC.

ACKNOWLEDGMENT

Dr. Olson would like to thank E. Francis Cook, Garret

Fitzmaurice, Tobias Kurth, and John Seeger from the Harvard

School of Public Health for their instruction in regression modeling

and propensity score analysis.

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Pediatr Blood Cancer DOI 10.1002/pbc

TABLE V. Relationship Between Patient Variables and Quality of Life

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Quality of life score

If yes (s.e.) If no (s.e.) P-Value

Legal difficulties 79.96 (3.82) 91.83 (2.10) 0.005

Diagnosis age< 5 86.48 (3.31) 88.02 (2.09) 0.682

CNS tumor 83.71 (3.47) 89.25 (2.02) 0.147

Chemotherapy 88.92 (2.00) 82.19 (4.15) 0.113

RT 87.09 (1.88) 94.80 (5.38) 0.318

CNS RT 87.45 (2.14) 89.48 (2.75) 0.605

Female 82.95 (2.92) 91.89 (1.85) 0.011

Relapse 93.10 (2.84) 86.74 (1.97) 0.256

Secondary tumor 87.38 (3.94) 87.49 (1.98) 0.984

CNS, central nervous system; RT, Radiotherapy; s.e., standard error.

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