Original Research

Cancer Screening for Dermatomyositis: A Survey of Indirect Costs, Burden, and Patient Willingness to Pay

Author and Disclosure Information

Dermatomyositis (DM) is a rare idiopathic inflammatory myopathy (IIM) associated with an increased risk for malignancy. Although cancer screening is recommended, no consensus guidelines currently exist. Whole-body positron emission tomography/computed tomography (PET/CT) has similar cost and efficacy to a more traditional conventional cancer screening panel (CSP). Our study sought to characterize patients’ perspective of cancer screening and the indirect costs to patients. We conducted a survey of patients recently diagnosed with DM who were undergoing or had recently undergone a CSP. Patient values and indirect costs need to be considered in choosing a screening modality. This study contributes to a greater understanding of patients’ experience of cancer screening in DM, which should be taken into consideration when developing consensus guidelines for cancer screening.

Practice Points

  • Dermatomyositis (DM) is associated with an increased risk for malignancy. Patient perspective needs to be considered in developing cancer screening guidelines for patients with DM, particularly given the similar efficacy of available screening modalities.
  • Current modalities for cancer screening in DM include whole-body positron emission tomography/computed tomography (PET/CT) and a conventional cancer screening panel (CSP), which includes a battery of tests typically requiring multiple visits. Patients may find the simplicity of PET/CT more preferrable than the more complex CSP.
  • Indirect costs of cancer screening include missed work, travel and childcare expenses, and lost wages. Conventional cancer screening has greater indirect costs than PET/CT.



Dermatomyositis (DM) is an uncommon idiopathic inflammatory myopathy (IIM) characterized by muscle inflammation; proximal muscle weakness; and dermatologic findings, such as the heliotrope eruption and Gottron papules.1-3 Dermatomyositis is associated with an increased malignancy risk compared to other IIMs, with a 13% to 42% lifetime risk for malignancy development.4,5 The incidence for malignancy peaks during the first year following diagnosis and falls gradually over 5 years but remains increased compared to the general population.6-11 Adenocarcinoma represents the majority of cancers associated with DM, particularly of the ovaries, lungs, breasts, gastrointestinal tract, pancreas, bladder, and prostate. The lymphatic system (non-Hodgkin lymphoma) also is overrepresented among cancers in DM.12

Because of the increased malignancy risk and cancer-related mortality in patients with DM, cancer screening generally is recommended following diagnosis.13,14 However, consensus guidelines for screening modalities and frequency currently do not exist, resulting in widely varying practice patterns.15 Some experts advocate for a conventional cancer screening panel (CSP), as summarized in Table 1.15-18 These tests may be repeated annually for 3 to 5 years following the diagnosis of DM. Although the use of myositis-specific antibodies (MSAs) recently has helped to risk-stratify DM patients, up to half of patients are MSA negative,19 and broad malignancy screening remains essential. Individualized discussions with patients about their risk factors, screening options, and risks and benefits of screening also are strongly encouraged.19-22 Studies of the direct costs and effectiveness of streamlined screening with positron emission tomography/computed tomography (PET/CT) compared with a CSP have shown similar efficacy and lower out-of-pocket costs for patients receiving PET/CT imaging.16-18

Conventional Cancer Screening Panel for Dermatomyositis

The goal of our study was to further characterize patients’ perspectives and experience of cancer screening in DM as well as indirect costs, both of which must be taken into consideration when developing consensus guidelines for DM malignancy screening. Inclusion of patient voice is essential given the similar efficacy of both screening methods. We assessed the indirect costs (eg, travel, lost work or wages, childcare) of a CSP in patients with DM. We theorized that the large quantity of tests involved in a CSP, which are performed at various locations on multiple days over the course of several years, may have substantial costs to patients beyond the co-pay and deductible. We also sought to measure patients’ perception of the burden associated with an annual CSP, which we defined to participants as the inconvenience or unpleasantness experienced by the patient, compared with an annual whole-body PET/CT. Finally, we examined the relative value of these screening methods to patients using a willingness-to-pay (WTP) analysis.

Materials and Methods

Patient Eligibility—Our study included Penn State Health (Hershey, Pennsylvania) patients 18 years or older with a recent diagnosis of DM—International Classification of Diseases, Ninth Revision code 710.3 or International Classification of Diseases, Tenth Revision codes M33.10 or M33.90—who were undergoing or had recently completed a CSP. Patients were excluded from the study if they had a concurrent or preceding diagnosis of malignancy (excluding nonmelanoma skin cancers) or had another IIM. The institutional review board at Penn State Health College of Medicine approved the study. Data for all patients were prospectively obtained.

Survey Design—A survey was generated to assess the burden and indirect costs associated with a CSP, which was modified from work done by Tchuenche et al23 and Teni et al.24 Focus groups were held in 2018 and 2019 with patients who met our inclusion criteria with the purpose of refining the survey instrument based on patient input. A summary explanation of research was provided to all participants, and informed consent was obtained. Patients were compensated for their time for focus groups. Audio of each focus group was then transcribed and analyzed for common themes. Following focus group feedback, a finalized survey was generated for assessing burden and indirect costs (survey instrument provided in the Supplementary Information). REDCap (Vanderbilt University), a secure web application, was used to construct the finalized survey and to collect and manage data.25

Patients who fit our inclusion criteria were identified and recruited in multiple ways. Patients with appointments at the Penn State Milton S. Hershey Medical Center Department of Dermatology were presented with the opportunity to participate, Penn State Health records with the appropriate billing codes were collected and patients were contacted, and an advertisement for the study was posted on StudyFinder. Surveys constructed on REDCap were then sent electronically to patients who agreed to participate in the study. A second summary explanation of research was included on the first page of the survey to describe the process.

The survey had 3 main sections. The first section collected demographic information. In the second section, we surveyed patients regarding the various aspects of a CSP that focus groups identified as burdensome. In addition, patients were asked to compare their feelings regarding an annual CSP vs whole-body PET/CT for a 3-year period utilizing a rating scale of strongly disagree, somewhat disagree, somewhat agree, and strongly agree. This section also included a willingness-to-pay (WTP) analysis for each modality. We defined WTP as the maximum out-of-pocket cost that the patient would be willing to pay to receive testing, which was measured in a hypothetical scenario where neither whole-body PET/CT nor CSP was covered by insurance.26 Although WTP may be influenced by external factors such as patient income, it can serve as a numerical measure of how much the patient values each service. Furthermore, these external factors become less relevant when comparing the relative value of 2 separate tests, as such factors apply equally in both scenarios. In the third section of the survey, patients were queried regarding various indirect costs associated with a CSP. Descriptions for a CSP and whole-body PET/CT, including risks and benefits, were provided to allow patients to make informed decisions.


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