Caregivers' perceptions of medical compliance in adolescents with cancer

J O U R N A L OF A D O L E S C E N T H E A L T H CARE 1986;7:22-27

Caregivers' Perceptions of Medical Compliance in Adolescents with Cancer MICHAEL J. D O L G I N , P h . D . , ERNEST R, KATZ, P h . D . , SHELLEY R. D O C T O R S , P h . D . , A N D STUART E. SIEGEL, M , D .

Two Studies were conducted to investigate the preValence and determinants of gross medical regimen noncompliance among adolescent cancer patients. In the first study, 28 children and adolescents were rated by their primary physicians on various aspects of medical regimen compliance, features of their disease, and treatment reg imens. Adolescent patients overall were judged to be significantly less compliant than the younger patients (p < 0.001), with almost half rated as being "'poor, or "very poor" compliers. Poor compliance was judged to be a potential threat to the prognoses of over half of the adolescents Studied. Adolescents were reported to experience more severe Side-effects and more visible physical residua as a result of their illness and treatment than the younger patients. The second study, con(iucted at a large pediatric referral center, found far fewer compliance problems among the 65 adolescent patients reviewed, with negative ramifications for treatment outcome reported in only nine patients (14%). The impact of medical variables, patient characteristics, and treatment setting on compliance with antineoplastic therapy is discussed. Issues in compliance assessment and considerations for future research are addressed. It is concluded that routine assessment of compliance is Crucial for monitoririg patient behavior as well as for the reliable evaluation of treatment protocols. KEY WORDS:

Medical compliance Cancer From the Division of Hematology-Oncology, Childrens Hospital of Los Angele's; the Division Of Adolescent Medicine, Montefiore Hospital Medical Cenier, New York; the Department of Pediatrics, University of Southern California School of Medicine; and the Department of Pediati'ics, Albert Einstein College of Medicine. Address reprint requests to" Michael J. Dolgin, Ph.D., Psychosocial Program, Division of Hemqtology-Oncology, Childrens Hospital of Los Angeles, P.O. Box 54700, Los Angeles, CA 90054. Manuscript accepted February 4, 1985. 22 0197-0070/86/$3.50

Noncompliance with medical regimens is a pervasive problem in health care, with potentially lifethreatening consequences in diseases such as caricer (1-3). Judging by the minimal attention it has received in the ~cientific literature, compliance with pediatric antic~incer regimens appears to have beeri largely taken for granted (4). Based on the complex therapeutic regimens used in cancer treatment and the important relationshi p between compliance and treatment outcome, a critical examination of the problem is warranted. Adolescent Coping with the realities of malignant disease and compliance with the demands of treatment must be viewed in the context of normal adolescent development (5). This is particularly true because advances in the medical managemen t of childhood and adolescent cancer have led to a shift in psychosocial emphasis from death and bereavement to living With a chronic disease and its treatment (6,7). The diagnosis and treatment of a serious illness during adOlescence threatens the attainment Of basic psychos0cial developmental tasks (5,8). The adolescent's Sense of personal autonomy is compromised by hospitalization and frequent clinic visits, which shift control over the adolescent's life to the instituti0n and its staff. The disease and treatment side-effects may cause physical weakness and debilitation; tending to make the patient dependent on others for even the most basic of needs. Parents may complicate matters by becoming over-protective--often as a means of combating their own feetings of fear and helplessness. Psychosocial and psychOsexual development may be impeded due to the physical chariges caused by the illness and its treatment.

© Society for Adolescent Medicine, 1986 Published by Elsevier Science Publishing Co., Inc., 52 Vanderbilt Ave., New York, NY 10017

January 1986

Hair loss, weight gain, emaciation, skin discoloration, disfiguring surgery, and the effects of gonadal radiation are major obstacles to the maintainence of a positive body image, sexual identity, and self-concept. School functioning and peer relations may be disrupted, and withdrawal from normal activities is not unusual. The adolescent's future orientation is shrowded in uncertainty in terms of long-term survival, educational goals, employment opportunities, and prospects for fertility and parenthood (9). Several factors may promote noncompliance in some patients. Cancer is frequently considered to be an incurable disease and patients may not consider their chances for survival worth the ordeal of aggressive therapy. Alternatively, noncompliance may represent the denial of the illness and its severity (8). For the adolescent patient, noncompliance may also represent an attempt to maintain independence in a situation of enforced dependency (8). The degree to which this problem manifests itself depends on a delicate balance in which parents and caregivers allow for growth and control in other areas of the patient's life in addition to sensitively involving the patient in his or her medical care. The literature indicates that treatment regimens that are protracted and complex in nature tend to result in reduced compliance (2,3,10). Aversive medical procedures and treatment side-effects may also hinder compliance, especially as these continue while the disease is in remission and the patient may otherwise be feeling well (11). Research has shown that pediatric cancer patients do not habituate to painful medical procedures, and that anxiety may actually intensify as clinic visits and illness duration progress (12,13). Results of earlier studies are mixed regarding whether adolescents are generally less compliant with medical regimens than younger patients, although caregivers often report this to be the case (2,14). Smith et al. (15) evaluated prednisone compliance in 52 children and adolescents with acute leukemia or lymphoma and found a 33% noncompliance rate for the entire sample. Separate analysis of the adolescent subjects revealed a 59% noncompliance rate. In another study, Leventhal and Boeck (16) reported treatment refusal and/or frequently missed appointments in ten of their 17 adolescent patients. In addition to depriving patients of improved health status, poor compliance also precludes the reliable assessment of treatment effectiveness (4). This may be particularly relevant as adolescents with acute leukemia tend to have poorer prognoses than

CAREGIVER'SPERCEPTIONS

23

younger children with the same disease (17). As Smith et al. (15) point out, patients treated with the same regimen have shown different induction rates and remission lengths. "Data collected at one institution are not always reproducible by a cooperative group (despite similar protocols) . . . . One of the factors that may influence these conflicting results is poor patient compliance" (p. 172). We conducted two studies to assess the scope and determinants of gross noncompliance in adolescent cancer patients. Gross noncompliance refers to factors such as missed appointments, treatment resistance or refusal, treatment discontinuation against medical advice, and noncompliance with follow-up requirements.

Methods Study 1

Subjects. All current patients in the pediatric hematology-oncology registry at Montefiore Hospital and Medical Center (MHMC), New York were eligible for inclusion in the study. However, patients diagnosed prior to 1979 and those who had completed their treatment course were excluded. The study sample consisted of 14 male and 14 female patients aged 3-18 years (mean = 10.3 years). Seventeen patients were ~ 12 years old. Sixteen patients had acute leukemia, four had lymphoma, and eight had a variety of solid tumors including neuroblastoma, rhabdomyosarcoma, teratoma, osteogenic sarcoma, and medullablastoma. Consistent with the epidemiological distribution of pediatric malignancies, acute leukemia was most common among the younger patients, while lymphomas and solid tumors were predominant in the adolescent group. Measures. A Caregiver's Questionnaire was designed to collect information regarding the characteristics of the disease, details of the treatment regimen, and his or her perception of the patient's compliance. Primary attending physicians completed these questionnaires, which covered the following: 1. diagnosis and probability of five-year survival at time of diagnosis and at the time of the study, 2. description of the treatment regimen, including duration, specific requirements (e.g., surgery, radiation therapy, chemotherapy, hospitalization,

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DOLGINET AL.

out-patient clinic visits), and a rating of the overall complexity of the treatment protocol based on its specific features and demands, 3. treatment side-effects, including the occurrence and severity of 14 common side-effects of antineoplastic therapy, visibility of physical disfigurement/impairment, long-term sequelae of treatment, and a rating of the overall severity of these side-effects, 4. patient compliance, including specific manifestations of gross noncompliance (e.g., missed appointments, treatment resistance or refusal, attrition from treatment or follow-up), a rating of overall patient compliance, and the potential impact of the patient's compliance on treatment outcome. The reliability of the Caregiver's Questionnaire was assessed by having physicians independently rate those patients with w h o m they were well acquainted. Agreement was obtained for 80% of all items, yielding an interrater reliability coefficient of r = 0.90. Because of the potential subjectivity involved in caregivers" ratings of patient compliance, further validation of the questionnaire was accomplished by a blind review of all in- and out-patient medical charts and progress notes. The review focused on indicators of gross noncompliance. Each patient was assigned an independent compliance rating ranging from I (very good) to 5 (very poor). These ratings and the caregivers ratings of overall patient compliance for the entire study sample (n = 28) yielded a Spearman correlation coefficient of 0.81 (p = 0.01), supporting the validity of caregivers' ratings as a measure of gross noncompliance.

Study 2 Subjects. Sixty-five adolescent cancer patients, aged 12-18 years (mean = 14.2 years) w h o were under active treatment at Childrens Hospital of Los Angeles (CHLA) were studied. Thirty-eight patients were male and 27 were female. Diagnoses included 39 hematologic malignancies and 26 solid tumors.

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Results Study 1 Caregivers ratings of overall patient compliance are presented in Table 1. While all of the younger patients received compliance ratings of "good" or "very good," only about one-third of the adolescent patients received such ratings. Almost half (45.5%) of the adolescent patients received ratings of "poor" or "very poor." Mean differences in individual compliance items revealed adolescent patients to be significantly less compliant as determined by missed appointments, treatment resistance or refusal, treatment discontinuation against medical advice, and nonadherence to follow-up requirements. Physicians were asked to rate the degree to which, in their judgement, noncompliance might influence treatment outcome. Noncompliance was judged to be of at least possible detriment to prognosis in 6 of the 11 (54.6%) adolescents studied. In the remaining five cases, noncompliance was not expected to interfere with the therapeutic outcome. Because of their generally good compliance, the prognoses of all of the younger patients was judged to be unaffected. Several treatment-related variables distinguished between the two compliance groups. As Table 2 shows, the adolescents were judged to experience significantly more severe side-effects as compared to the younger patients, specifically in terms of nausea and vomiting, anorexia, mouth and skin discoloration, and extremity pain or weakness. Treatmentrelated disfiguration and physical residua were significantly more evident among adolescent patients. Treatment regimens prescribed for adolescents were judged to be more complex than for younger patients, although this difference did not reach statistical significance. Table 1. Caregivers' Ratings of Overall Compliance (Study 1) Compliance ratings Very poor

Poor

Fair

Very G o o d good

(n) (%) (n) (%) (n) (%) (n) (%) (n) (%)

Measures. Primary attending physicians were asked to rate each patient's compliance using the Caregivers Questionnaire described above. Specific items focused on gross noncompliance, potential impact of noncompliance on treatment outcome, and caregivers' perceptions of the barriers to patient compliance.

Children aged 411 years (n = 17) Adolescents aged 12-18 years (n = 11) Total (n = 28)

0

0

0

0

0

0

4 23.5 13 76.5

2 18.2 3 27.2 2 18.2 2 18.2 2 18.2 2 7.1 3 10.7 2 7.1 6 21.4 15 53.6

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CAREGIVER'SPERCEPTIONS

Table 2. Treatment-related Differences Between Children and Adolescents (Study 1)

Item Nausea and vomitinga Anorexiaa Skin discolorationa Pain/weaknessa Mouth/lip ulcersa Physical residua a Overall severity of sideeffectsb Overall complexity of regimenc

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Table 3. Caregivers' Survey (Study 2)

Children (n = 17) ~

Adolescents (n = 11) 2

p

1.41 0.76 0.41 0.76 0.75 0.53

2.09 1.55 1.20 1.45 1.73 1.09

<0.01 <0.01 <0.05 <0.05 <0.01 <0.05

2.47

3.18

<0.05

2.70

3.27

N.S.

(n)

a0 = none; 1 = mild; 2 = moderate; 3 = severe. bl = very mild; 5 = very severe. cl = very simple; 5 = very complex. Several illness- a n d treatment-related variables were correlated w i t h compliance for the entire sample (n = 28). Poor compliance was associated with more severe t r e a t m e n t side-effects (r = -0.40; p < 0.05) and more visible physical residua resulting from the illness a n d t r e a t m e n t (r = -0.38; p < 0.05). More complex regimens t e n d e d to be related to poor compliance (r = -0.30), a l t h o u g h this was not statistically significant. Prognosis at diagnosis a n d treatm e n t duration were positively correlated w i t h compliance for the sample as a whole (r = 0.50; p < 0.01 and r = 0.38; p < 0.05, respectively), possibly because the generally more compliant y o u n g e r children t e n d e d to have more favorable prognoses a n d more protracted regimens. W h e n adolescent patients were considered alone (n = 11), compliance was negatively correlated w i t h t r e a t m e n t duration (r = -0.33), a l t h o u g h it was n o t statistically significant due to the small sample size. Study 2 Table 3 s u m m a r i z e s the responses to the Caregivers Questionnaire. Over 80% of the adolescent patients at C H L A were rated as " g o o d " or "'very g o o d " cornpliers, with significant compliance difficulties reported in approximately one in ten patients. Noncompliance was j u d g e d to be of potential d e t r i m e n t to treatment o u t c o m e in only nine cases (14%). In those instances where compliance problems were reported, caregivers most c o m m o n l y attributed these problems to the t r e a t m e n t side-effects, poor family and social support, denial of the illness' severity, a n d lack of belief in treatment efficacy.

Compliance ratings Very good 43 Good 10 Fair 5 Poor 3 Very poor 4 Impact on prognosis None 56 Possible 3 Certain 6

(%) 66 15 8 5 6 86 5 9

Discussion The major findings of S t u d y 1 concerned the high degree of gross noncompliance a m o n g adolescent patients a n d the potential impact of their behavior on prognosis, as j u d g e d by their primary physicians. Results of earlier studies differ as to w h e t h e r adolescents are actually less compliant t h a n y o u n g e r patients, h o w e v e r our findings s u p p o r t this notion in adolescent cancer patients (2,14). Our results corroborate those of Smith et al. (15) w h o f o u n d poorer medication compliance a m o n g the adolescent patients studied. Prognosis at diagnosis, severity of side-effects, and obvious physical residua were significantly related to noncompliance in Study 1. Leventhal a n d Boeck (16) discussed these factors, particularly fear of discomfort a n d deformity, as major contributors to depression a n d noncompliance in adolescents with cancer. In this sense, noncompliance m a y represent an attempt at disease denial which, despite its adaptive a n d self-preserving functions, m a y pose a serious threat to the patient's care (18). Physicians" perceptions of barriers to compliance in S t u d y 2 support this formulation, with side-effects of treatment a n d denial of the illness a n d its severity j u d g e d to be major limiting factors to compliance. In Study 2, physicians reported significant compliance problems in about one in ten adolescent patients. These results suggest a more optimisitc view of gross adolescent compliance t h a n the findings of our first study. Several factors might account for these divergent findings. O n e factor involves the treatment setting. S t u d y I was c o n d u c t e d at a general inner-city hospital w i t h a relatively small pediatric oncology service. The patients studied were primarily comprised of residents in the immediate catchment area served by M H M C . M a n y were so-

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DOLGINET AL.

cioeconomically disadvantaged and thus were confronting a variety of stressors in addition to their illness. Some of these patients had previously been treated at a large cancer treatment center, but were referred back to the local facility because of compliance problems in the hope that closer proximity to the health care setting might improve compliance. In contrast, major pediatric cancer referral centers such as CHLA attract patients from much greater distances in addition to local patients. These patients often seek care at a major center over the smaller facilities closer to home. Thus, different motivating factors in the pursuit of treatment may account for some of the differences in patient compliance observed in these two settings. A second possibility relates to the availability of routine psychosocial support and follow-up at the two study centers. Comprehensive psychosocial care (i.e., routine involvement of psychologists, social workers, and patient activity specialists) was available to patients at CHLA and this might have reduced the extent of adolescent noncompliance. The interaction of treatment setting and patient compliance is complex and poorly understood and constitutes an important area for future investigation. Pediatric as well as adult cancer patients are treated in a variety of settings; therefore, patient management problems and solutions may not be generalizable from one type of setting to the next. Future research must also explore and refine appropriate methods for assessing patient compliance in this population. Caregivers' estimates of patient compliance, the primary data source for our studies, have been criticized because of their subjective nature (2,3,19). This criticism however has been primarily leveled at caregivers' estimates of home medication compliance and other indicators to which the physician has no direct access. In contrast, our focus was on gross noncompliant behaviors to which the caregiver has direct and primary access. Furthermore, caregivers' judgements have been demonstrated to underestimate noncompliance, rendering our present findings conservative (20). Given our findings on gross noncompliance, it is likely that medication compliance and preventative health measures are even more seriously deficient among adolescent patients. More sophisticated methods are needed to routinely monitor compliance. This is exemplified by the Smith et al. (15) study of prednisone compliance; however, even such direct biochemical measures may be of questionable reliability or validity due to individual differences in absorption rate, metabolism, or drug interactions. Multiple prospec-

JOURNALOF ADOLESCENTHEALTHCAREVol. 7, No. 1

tive measures will ultimately provide the most accurate information on compliance, and future research should move in that direction. Knowledge of the factors associated with good and poor compliance in children and adolescents with cancer is limited, despite the fact that such data would facilitate the early identification of high-risk patients and foster the development of appropriate intervention strategies. Lansky et al. (21) found compliance with oral prednisone in patients under 15 years of age to be more closely related to parent than to patient characteristics. For adolescent patients, attempts to understand the factors associated with compliance have been reported on a single case study basis. Smith et al. (22) describe a case in which the patient's low anxiety, borderline intelligence, hostility, and external locus of control were deemed responsible for his poor compliance. The relative contributions of patient characteristics and parental involvement may vary in determining the levels of compliance observed in children and adolescents. Parental supervision and control over the preschool or school-aged child may be greater than that exercised over the adolescent. Adolescents have and strive for greater autonomy than younger children in health and illness behavior as well as in general decision making. This may explain the age-related differences in compliance found in Study 1. Even among adolescent patients, however, both patient and parent factors are crucial. Results of our in-depth study of eight adolescents suggest several different patterns of noncompliance that can be grossly classified into two categories: parent-initiated and patient-initiated (23). The relationship between compliance and treatment outcome is an area of major importance (1,24). Is noncompliance associated with higher rates of morbidity and mortality or do patients w h o receive less than maximum therapy achieve treatment outcomes comparable to those of compliant patients? Our studies suggest that in the subjective eyes of caregivers, such an association does appear to exist. Objective outcome data are needed. Results of an initial two-year follow-up study of six noncompliant adolescent cancer patients suggests a higher rate of disease recurrence and mortality w h e n compared to a group of compliant adolescent patients matched for tumor site, stage, and treatment protocol (Dolgin, Katz, and Siegel, unpublished). It may be beneficial to include gross as well as indirect measures of patient compliance as part of routine care, particularly for patients on investigational protocols. Without such data, definitive conclusions regarding the effec-

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CAREGIVER'S PERCEPTIONS

tiveness or ineffectiveness of specific regimens may be premature. 12.

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