Rheumatic diseases affect an estimated 300,000 children in the United States. Despite this large number of affected children, a severe shortage of pediatric rheumatologists to diagnose and manage these patients remains. These workforce issues result in delayed diagnosis and treatment, which impedes efforts to achieve the best outcomes, which we now know result from early and effective treatment [18–20]. Telemedicine has the potential to overcome the barriers of time and distance for families and improve access to pediatric rheumatologists.
Our results are consistent with previous estimates of the average distance travelled to see a pediatric rheumatologist. The American Academy of Pediatrics estimates that about ¼ of children with rheumatic disease live 80 miles or more from a pediatric rheumatologist, which is in line with the findings in our study [21]. The median distance travelled by a family to the Kansas City clinic was 40 miles and one-quarter of our patients travelled at least 85 miles for the clinic visit. Several other pediatric subspecialists in addition to pediatric rheumatology are faced with access issues. A study examining the supply and utilization of pediatric subspecialists for 13 chronic medical conditions, one of which was arthritis, found that when parents in the lowest quintile of subspecialist supply reported an unmet need for subspecialty care, the most common barriers were related to lack of provider in the area or transportation concerns. Additionally, among all children with chronic conditions, a significantly larger percentage of children with JIA lived in areas in the lowest quintile of subspecialist supply as compared to areas in the highest quintile of supply [22]. If children with chronic arthritis are unevenly distributed, with greater numbers in areas with a decreased supply of pediatric rheumatologists, improvement in access to care for these children takes on even greater magnitude.
Because individuals with known rheumatic disease are required to make frequent visits to a rheumatologist, travelling such distances can result in the accumulation of substantial personal costs to a patient and family over time. Studies investigating the economic burden of pediatric rheumatic disease are limited, and primarily pertain to juvenile idiopathic arthritis (JIA), which is the most common rheumatic disease seen in pediatric rheumatology clinics. Results of studies are varied due to insurance differences among countries, inconsistent inclusion of patient non-health care costs, and the increasing use of biologics, which are more costly. Compared to children seen in an outpatient clinic without JIA, those with JIA had substantially higher costs related to medication use, visits to specialists and other health care providers, and diagnostic tests [23]. A Turkish study found that medications, especially tumor necrosis factor-inhibitors, accounted for about 85 % of total patient costs [24]. However, when evaluating children who were not on tumor necrosis factor-inhibitors, transportation and lodging expenses contributed to 35 % of total costs. A study conducted in Germany showed different results; transportation costs composed a majority of the out-of-pocket costs per year for a family [25]. In this cohort, 23 % of mothers and/or fathers had missed work related to their child’s JIA. This is in stark contrast to our cohort where 64 % of respondents reported missing work to attend their child’s appointment. A Nova Scotia study evaluated both patient costs as well as perceived financial burden of JIA for families [26]. Non-medical costs, specifically costs associated with visits to the tertiary care center where the rheumatology clinic was located, composed 31 % of the total costs. Annual loss of paid work accounted for another 33 % of total annual costs. Notably, the perceived financial burden of JIA was rated as either large or moderate by 36 % of the respondents and 36 % of the respondents also felt that resources to assist with costs were poor. The authors noted that even though the overall costs of having a child with JIA were modest in this study, many families still perceived the financial burden as significant and access to resources as poor.
Currently there are no published studies to our knowledge investigating the financial impact on families who utilize telemedicine in pediatric rheumatology. Even in systematic economic evaluations of telemedicine programs in general, there has been a focus on cost savings to the health care system, with less focus on financial benefits for patients. Although 29 states have mandated that commercial insurance cover telemedicine encounters, rates of reimbursement vary greatly. Despite much research indicating the efficacy of telemedicine and legislative progress, there are still reimbursement barriers related to where patients are located, what types of providers are considered eligible for reimbursement and what services are covered. The evaluation of telemedicine programs is complex and prior reviews of the literature have revealed a lack of high-quality, rigorous investigations [27, 28]. With the development of our Joplin telemedicine clinic, respondents were less likely to spend money on food, traveled a shorter distance to clinic and spent less time away from work and school compared to when they had traveled to Kansas City. Importantly, when the Joplin cohort was seen via telemedicine, this leveled the economic burden to where it was similar to the Kansas City cohort; there were no significant differences in the percentage of families who spent money on lodging, distance traveled to clinic, and the time missed from school and work between these two groups.
Despite the potential for telemedicine to improve access to care in pediatric rheumatology, it is imperative to also demonstrate that these children are receiving high quality care via this innovative method. Multiple studies in the adult rheumatology population show successful utilization of telemedicine for new patient consultations. A feasibility study in 52 new patients seen via teleconsultation where a general practitioner examined the patient while the rheumatologist observed resulted in high levels of satisfaction among the patient, general practitioner and rheumatologist. No patients required a face-to-face visit after the telemedicine consultation [29]. We have had similar satisfaction with our pediatric rheumatology telemedicine clinic. Although not part of the current study, routine surveys given to 36 patients and families at the end of rheumatology telemedicine clinic visits show that 100 % of participants would recommend a telemedicine visit to a family member or friend. When asked if visiting with the rheumatologist using telemedicine was just like the provider being in the room, 78 % strongly agreed and 22 % mostly agreed.
One of the major deficits in the tele-rheumatology literature is the lack of evidence regarding the ability to conduct a patient examination remotely and develop appropriate assessments for ongoing care [30]. A single non-randomized study of 100 new adult rheumatology referrals looked at the diagnostic accuracy in telemedicine visits. Diagnostic accuracy, which was defined as the percentage of equivalent diagnoses between a live, interactive telemedicine visit and an in-person consultation, was 97 % [31]. Notably, there have been no studies conducted in patients with known rheumatic diseases who must make frequent visits to the rheumatologist for their ongoing care, which can result in substantial financial burden to the patient over time. Certainly, the benefits of easy access are negated if the quality of care provided is subpar. Additional questions pertaining to best practices in telemedicine arise, including: how often do children with rheumatic diseases need to be seen by telemedicine, should telemedicine visits alternate with in-person visits, can the team-based approach that incorporates the services of multiple health care providers (e.g. rheumatologist, physical and occupational therapists, social worker, and psychologist) be delivered via telemedicine, and are children with certain specific rheumatic diseases better suited to be seen by telemedicine than those with other diseases? Future studies need to address these numerous questions. However, our results show that interest in this care delivery method exists and there is great potential in cost savings.
Our study has potential limitations inherent to all survey based studies. It is possible that respondents may have overestimated costs related to their rheumatology appointments. However, we do know that the reported distance to clinics by respondents is consistent with data obtained from prior studies [1, 21]. Additionally, the survey was changed during the course of the study to allow for open ended answers. This led to variable response rates among the questions depending on the ability to combine answers from different survey answers during the statistical analyses. The sample size of the Joplin telemedicine population was small, though a near 100 % response rate within this group decreases the risk of sampling error. Although this survey was conducted at a single center, which inherently limits generalizability, the clinic services four surrounding states and the population served is a mixture of rural, suburban and urban patients by the nature of the region. Finally, our study focused on time and cost savings for families related to telemedicine visits. These factors were based on our interest in these issues as drivers for interest in telemedicine. Certainly, other factors, such as money spent on clinic visits and medications, convenience or access to multidisciplinary care, insurance coverage for labs, radiology services or clinic visits themselves impact the care of a child with rheumatic disease.
