Forty six percent of unaffected, at-risk men from multiple-case TC families in our sample reported regular performance of TSE. The two factors significantly associated with regular performance of TSE were physician recommendation and higher levels of TC cancer worry.
In a 2005 review of TSE practices that included 6 studies of U.S. men ≥ age 18 [39
], the percent of men practicing TSE on a monthly basis ranged from 6 to 36% Our TSE rates are not directly comparable to those data, which defined regular performance of TSE as once a month, since we used a broader inclusion for our regular performer category of 6 times a year or more. This may explain the higher proportion of men in our sample that were defined as regular performers of TSE (46%).
With respect to the HBM variables assessed in the current study, only Cues to Action (i.e. physician recommendation for TSE), remained a statistically significant predictor of TSE behavior in multivariate analysis. The importance of physician recommendation on TSE practice was striking, with men who reported a physician recommendation for TSE having at least six times higher odds of performing regular TSE compared with those who reported no such recommendation. This provides yet another example [33
] of how potent an influence the physician can be with regard to cancer prevention behavior in patients, and underscores the importance of physicians including such recommendations in the course of ongoing health care discussions. Due to the lack of a strong evidence base to support the belief that TSE is associated with improved TC-related survival among those who practice it, and the remarkable therapeutic successes in managing TC patients even with far-advanced disease, it is unlikely that there ever will be a clinical trial testing this question. In this era of evidence-based medical practice, it is difficult for health care providers to insist that at-risk men adopt TSE practice, although common sense and logic suggest that a TC detected by TSE rather than symptoms should be more amenable to treatment regimens that do not include chemotherapy, or permit less intense or shorter chemotherapy regimens. This rationale is not unlike recommendations for both BSE, CA-125 testing, and transvaginal ultrasound recommendations for women at increased risk for hereditary breast and ovarian cancer [42
]. Given that TC is a disease which affects young men during the most productive period of their lives, there is potential for real economic and psychosocial benefit in attempting to minimize treatment-associated costs and morbidity.
Two previous studies of the HBM and TSE showed that perceived benefits and barriers were associated with TSE, as did our data [19
]. In our study, perceived cancer susceptibility was not associated with TSE. Although one previous study examining the relationship between TSE and perceived susceptibility found a significant association, there were only 12 men in the entire study who were considered to be "practicers" of TSE (i.e. reported practicing TSE > 4 times a year) [19
]. A larger study of TSE behaviors using multivariate analyses failed to replicate this finding [29
]. Perceived severity of TGCT was not a significant predictor of PSA testing. This finding is similar to two previous studies of TSE that used the HBM as a theoretical framework [19
]. Previous cancer screening research for a variety of sites has also found perceived susceptibility of little predictive value with regard to screening behaviors, possibly due to the almost universally held belief that cancer is a severe disease, regardless of type [43
]. In our study, none of the HBM factors significant in bivariate analyses remained so when they were considered in a multivariate model which included physician recommendation. This result is consistent with many previous studies of cancer screening behaviors.
To our knowledge only one other study has examined HBM with respect to TSE using multivariate analysis. Overall, it appears that the HBM does not adequately explain TSE behavior. Similar to our study, HBM variables accounted for only a small proportion of the variance in TSE behavior (21%) [29
]. As discussed below, cancer-related worry was the main psychosocial predictor of TSE behavior in our study and may lend some support to the commonly-raised concern regarding the HBM and other cognitive models of health behavior, namely that they do not include affective variables as behavior predictors [30
Cancer-related worry was the other major factor associated with TSE practice patterns in our study. From a theoretical perspective, several relationships between cancer worry and cancer prevention behaviors have been proposed. Some models posit that worry may serve as a facilitator to cancer screening behavior, others postulate worry acts a barrier, and some propose a curvilinear relationship between cancer worry and preventive behaviors, whereby worry is a facilitator to a certain level after which it becomes a barrier [49
]. The HBM, which was the theoretical basis of the present study, has been used to support the role of worry as both a barrier and facilitator for cancer screening behavior [49
]. In our sample, it appears that cancer worry motivates individuals to practice TSE, since those with higher levels of cancer worry were also more likely to perform TSE. However, the directionality of this relationship can only be established in the context of a prospective study; our data are cross-sectional. Studies in other high-risk populations provide some evidence that cancer worry may in fact precede
screening behavior. In a review of the relationship between cancer worry and mammography screening among high-risk women, four of five prospective
studies indicated that higher levels of cancer worry were associated with greater rates mammography screening and breast self-examination [49
]. However, no similar prospective studies of high-risk men are available. One cross-sectional study of prostate cancer screening among a sample of men at increased risk of hereditary prostate cancer found a negative relationship between cancer worry and screening behavior [50
It is noteworthy that we observed no statistically significant differences in TSE practices between men with and without a prior history of a testicular abnormality. A personal history of cryptorchidism can increase the risk of TC up to 11-fold [51
]. Of the 13 men reporting a testicular abnormality in our series, 29% (n = 4) reported a history of cryptorchidism (data not shown). In a study by Blesch and colleagues, none of the 5 men with a history of cryptorchidism, reported regularly practicing TSE [19
]. While this finding may reflect the small number of men with this particular risk factor and the associated lack of statistical power to detect a difference in that study, it is possible that men may be unaware that testicular maldescent is among the most strongly-established TC risk factors. However, no study to date has focused specifically on the levels of awareness/knowledge of cryptorchidism as a risk factor for TC or as a multivariate predictor of TSE behavior.
In multivariate analyses, men age = 35 and those who were first-degree relatives of a TC case were no more likely to perform TSE than were older men or those whose affected family member was a more distant relative. In the 2005 review mentioned above [27
], only 12–25% of participants in U.S. studies were aware of the age group most affected by TC. In the study by Blesch and colleagues [19
], men with a family history of TC were significantly more likely to perceive greater benefits to TSE. However, the study did not assess the impact on actual TSE behavior. Taken together, the non-differential rates of TSE between men in younger and older age groups, men who are first versus
second-degree relatives of a TC case, and men with a prior testicular abnormality compared with those without, suggest a lack of awareness on the part of unaffected family members regarding the additional risk conferred by having these characteristics. It is also possible that men recognize the implications of these characteristics, but they are not sufficiently concerned by this information to motivate screening behavior.
To our knowledge, this is the first study to examine TSE behaviors among unaffected men from multiple-case testicular cancer families. While the results provide important information about current rates and correlates of TSE in this high-risk group, they should be interpreted with caution due to certain limitations of our study. A goal of our study was to evaluate the utility of HBM in understanding TSE behavior. Ideally, use of an analytic technique such as structural equation modeling would allow us to definitively test the utility of HBM in predicting TSE behavior. However, our study was designed as a pilot study of the utility of HBM and it did not have a sample size sufficient to reach definitive conclusions. It is possible that the HBM did not adequately capture those factors that were important in TSE behavior. For example, one of the major criticism of HBM and other cognitive models of health behavior is the lack of inclusion of affective variables (e.g. distress) [30
]. To address this issue, we included scales for both cancer worry and cancer distress. Additionally, we included demographic and SES variables that may also impact screening behavior. Finally, in a study that compared the HBM to the Theory of Planned Behavior, the quality of the models was very similar. The TPB explained 22%of the variance in behavior versus
21% by the HBM, suggesting that applying another theoretical framework may have yielded similar results [29
]. Thus, based on our review of the literature related to theories of health behavior and predictors of screening, we feel that we adequately expanded the HBM to include other factors associated with TSE behavior. In addition, this was a cross-sectional study, making it difficult to establish that the beliefs and psychological factors we identified preceded the behavior, as hypothesized in the HBM. Additionally, men volunteering to participate in a study of familial TC may be more aware of and concerned about TC. Thus, the rates of TSE may be higher in this study population than men with a similar history not participating in such a study. Similarly, these men may be more knowledgeable about TC and have differing beliefs and psychological responses to TC.
As molecular genetic diagnosis improves, family history of TC will become an increasingly important risk factor on which to base decisions for primary, secondary, and tertiary prevention of TC. The present study showed that among men with a family history of TC, those who had higher levels of cancer worry were more likely to practice TSE regularly. However, findings also revealed that men at increased risk of TC due to a positive family history may be either less knowledgeable about and/or less motivated to perform TSE even if they are younger, have a prior testicular abnormality, or are a FDR of a man with TC. Thus, these are areas in which men may benefit from additional education and information upon which to base decisions about TSE practices. Physician recommendation emerged as the most important predictor of regular performance of TSE. Despite the absence of proof regarding the efficacy of TSE, it nonetheless remains prudent to encourage health care providers to recommend monthly TSE for high-risk patients, and to instruct their patients in performing this examination. Given the superficial/accessible location of the testes, and the existence of non-invasive imaging (testicular ultrasound) and tumor marker assays, the price one pays for a false-positive screening examination is likely to be substantially less than would be the case for ovarian cancer screening, for example. Finally, providing at-risk men information regarding accepted TC risk factors, as a means of deepening their understanding of their own risk, might help to correct some of the information deficiencies we identified. Despite the limitations in both our and other's studies, we have adopted the policy of recommending monthly TSE as a screening modality for unaffected bloodline males from multiple-case TC families, while clearly informing our patients that this represents our best clinical judgment rather than an evidence-based recommendation.