Co-chairs: Elizabeth Maloney and Rebecca Bunnell
Information and opinions in this report do not necessarily reflect the opinions of the working group, the U.S. Department of Health and Human Services, or any other component of the Federal Government. Readers should not consider the report or any part of it to be guidance or instruction regarding the diagnosis, care, or treatment of tick-borne diseases or to supersede in any way existing guidance. All subcommittee members actively participated in the development of this report. Members voted to approve submission of the report to the Working Group and on the wording of each of the potential actions contained in the report. The vote to submit the report indicates general agreement with the content of the document, but it does not necessarily indicate complete agreement with each and every statement in the full report.
Background
Health equity, in which patients with tick-borne diseases (TBDs) can reach their full health potential unburdened by structural and societal constraints, is the priority for the Access to Care and Education Subcommittee.
What Is Health Equity?
Health equity, as defined by the American Medical Association “is optimal health for all” (American Medical Association, 2018). It seeks “to continually reduce the burden of illness, injury, and disability, and to improve the health and functioning of the people of the United States” (Institute of Medicine, 2001). Health equity is a matter of fairness and justice; groups experiencing disparities are often marginalized or lack political, social, or economic power (American Medical Association, 2018; Liburd et al., 2020).
Why Is Health Equity Important?
“The ultimate test of the quality of health care is whether it helps the people it intends to help” (Institute of Medicine, 2001). Health care can only do so by ensuring access to necessary clinical expertise (Institute of Medicine, 2001). According to the Centers for Disease Control and Prevention (CDC), healthcare equity is achieved only “when every person has the opportunity to attain his or her full health potential and when no one is “disadvantaged from achieving this potential because of social position or other socially determined circumstances” (Centers for Disease Control and Prevention, 2020).
Equity, specifically health equity, has eluded many patients with TBDs. Health disparities—differences in access to care, longevity, rates of disease, disease severity, disability, quality of life, and death (Centers for Disease Control and Prevention, 2020)—are the result of structural and societal determinants of health inequity. Structural and social determinants of health act systematically through “long causal chains of mediating factors” (Solar & Irwin, 2010). Institutions, policies, and practices, including governmental policies and practices, that determine the distribution of power, funding, and the availability of services are structural determinants of health. Social determinants result from structural determinants and act within the context in which people live their lives; of the five broad categories of social determinants (Office of Disease Prevention and Health Promotion, n.d.-c), access to quality health care is the most critical for patients with TBDs.
The United States, as a whole, benefits from the well-being of its individual communities (Office of Disease Prevention and Health Promotion, n.d.-b). Yet, many patients with one or more TBDs face daunting health inequities such as:
- Decreased quality of life (Johnson et al., 2014; Rebman et al., 2017),
- Increased rates of disease (Fallon et al., 2021),
- Increased disease severity (Dennison et al., 2019),
- Preventable death (Congressionally Directed Medical Research Programs, 2022; Dahlgren et al., 2012; Marx et al., 2020), and
- Limited access to care (Johnson et al., 2011).
Of these, resolving limitations to accessing quality care could substantially reduce the other health inequities. Access to health care is “the timely use of personal health services to achieve the best health outcomes” (Institute of Medicine, 1993). This definition relies on both the availability and use of health services, as well as the quality of health outcomes, to determine whether access has been achieved.
Barriers to accessing quality health care include prohibitive costs and insurance coverage limitations, limited availability of services, and physical or geographic barriers. The inability to access care on a timely basis and the lack of capable, qualified, and culturally competent providers are also barriers to accessing quality health care (Office of Disease Prevention and Health Promotion, n.d.-a). Access to care may be affected by institutional and structural healthcare barriers as well as by hidden barriers, such as non-physician gatekeepers, and stigma, disrespect, or discrimination associated with medical care (Nyblade et al., 2019).
Executive Order 13985 calls for assessment tools that will allow agencies to determine whether “their policies and actions create or exacerbate barriers to full and equal participation by all eligible individuals” (Executive Office of the President, 2021). In light of the health inequities noted above, this subcommittee report identifies structural governmental barriers that impede patients from obtaining access to quality health care and that deter physicians from providing such care.
The Purpose of This Report
The goals of this report are to highlight existing inequities experienced by patients with TBDs, to focus attention on the government policies and processes that are major root causes of these inequities, and to propose actionable solutions to improve the health of this marginalized and often-neglected patient community.
Methods
See Appendix 1.
The subcommittee heard from the following presenters:
- Holly Ahern, MS, MT(ASCP), Associate Professor of Microbiology, State University of New York, Adirondack;
- Angel Davey, PhD, Program Manager, Tick-Borne Disease Research Program, Congressionally Directed Medical Research Programs;
- Gordon Guyatt, MD, Professor of Health Research Methods, McMaster University, and key developer of GRADE;
- Miranda Lynch-Smith, Deputy Assistant Secretary for Human Services Policy, Office of the Assistant Secretary for Planning and Evaluation, U.S. Department of Health and Human Services;
- Ruth Martinez, MA, CMBE; Executive Director, Minnesota Board of Medical Practice;
- Mary Nix, MS, Deputy Director, Division of Practice Improvement, Center for Evidence and Practice Improvement, Agency for Healthcare Research and Quality;
- Sue Partridge, MPH, CDR U.S. Public Health Service, Associate Director for Communication, Division of Vector-Borne Disease (DVBD), CDC, with Paul Mead, MD, MPH, Chief, Bacterial Disease Branch, DVBD, National Center for Emerging and Zoonotic Infectious Diseases, CDC
- Sam Perdue, PhD, Program Officer for Rickettsial and Related Diseases, National Institute of Allergy and Infectious Disease (NIAID);
- Jeremy Richards, MD, Chair, Department of Medical Education at Mount Auburn Hospital, and a medical educator at Harvard Medical School;
- Ginger Savely, DNP, tick-borne diseases clinician; and
- Richard Schwartzstein, MD, Ellen and Melvin Gordon Professor of Medicine and Medical Education, Harvard Medical School, and Director, Shapiro Institute for Education and Research, Harvard Medical School and Beth Israel Deaconess Medical Center.
Results and Findings
For consideration by the Tick-Borne Disease Working Group, the Access to Care and Education Subcommittee has identified one major priority and seven potential actions to achieve it.
Priority 1. To ensure health equity for patients with tick-borne diseases (TBDs) so that they may reach their full health potential unburdened by structural and societal constraints.
Background
Health inequity in TBDs has many origins, some of which are addressed in detail in previous Tick-Borne Disease Working Group reports (Tick-Borne Disease Working Group, 2018a, 2020b). Therefore, the following statement from the National Academy of Medicine is especially germane:
“The primary goals of medicine include improving health by providing beneficial care to patients, conducting valid research, and offering excellent medical education.” (Institute of Medicine, 2009)
This subcommittee’s work focuses on three key, structural, interwoven factors that account for many of the health disparities that patients with tick-borne illnesses experience:
- Patient-encountered barriers,
- Clinician-encountered barriers, and
- Medical educational barriers.
This report describes the major challenges and opportunities for each identified factor in order to demonstrate that these three factors share a common root cause as well as several shared solutions. The report primarily addresses barriers pertaining to patients with persistent Lyme disease/chronic Lyme disease (PLD/CLD). For the purposes of this report, patients who have been diagnosed with Lyme disease by a clinician and who remain ill for six or more months following antibiotic treatment are referred to as having PLD/CLD. The subcommittee chose to focus on the PLD/CLD group because the health inequities and barriers experienced by these patients are well documented. However, the subcommittee recognizes that patients with other TBDs face some of the same barriers, especially with regard to clinician knowledge gaps.
Summary of Evidence and Findings
Patient-Encountered Barriers to Achieving Health Equity for Patients with Tick-Borne Diseases
Lyme disease and other TBDs are often poorly understood, under-recognized, and subject to treatment failures (Bobe et al., 2021; Jibbe et al., 2021; Renard & Ben Mamoun, 2021). In the United States, when Lyme disease is localized to the skin and promptly diagnosed and treated with three weeks of antibiotics, the majority of cases resolve without complications (Maloney, 2021). However, treatment failures ranging from 10 to 43% have been reported in this group (Aucott et al., 2022; Luger et al., 1995). Treatment failure rates are generally higher for patients presenting with delayed diagnosis and late manifestations (Hirsch et al., 2020; Logigian et al., 1999; Steere & Angelis, 2006; Steere et al., 1994).
Patients with PLD/CLD are generally treated by clinicians who dedicate significant practice time caring for patients with PLD/CLD, family physicians, or rheumatologists; only 12% of patients report being treated by infectious disease specialists (Johnson et al., 2011). As such, there is a wide range of experiences among clinicians treating various manifestations/stages of Lyme disease. Clinicians who primarily care for acute manifestations of Lyme disease may be adept at recognizing them and prescribing initial treatment while missing complex presentations and patients with PLD/CLD. Similarly, clinicians who care for patients with advanced presentations or PLD/CLD may not appreciate that many patients are diagnosed early in the infection and successfully treated. These two clinician cohorts, whose knowledge of Lyme disease is based on the manifestations/disease state they typically treat, disagree with each other (Auwaerter, 2007; Maloney, 2016; Stricker, 2007). Although each cohort may be correctly reflecting its experiences and expertise, Lyme disease encompasses both disease states.
PLD/CLD patients report a substantial burden of illness (Rebman et al., 2017). In a survey of more than 3,000 PLD/CLD patients, 72% reported fair or poor health status, which significantly exceeds 16% in the general population (Johnson et al., 2014). Fifty-one percent of that survey’s respondents reported having PLD/CLD for more than 10 years. In another survey of more than 2,400 patients, 31% had applied for and 26% had received public support or received disability benefits (Johnson et al., 2011).
Health equity is a core issue in PLD/CLD, which is a marginalized and stigmatized illness with significant access to care barriers (Johnson, 2019b; Tick-Borne Disease Working Group, 2020b). Although health inequities are often associated with race, ethnicity, and sexual orientation, they also arise in the context of specific marginalized or stigmatized healthcare conditions such as mental illness, HIV/AIDS, substance abuse, hepatitis, and Lyme disease. As noted earlier, health inequities may be reflected in quality of life, disease severity, disability, morbidity, and access to care and treatment (Centers for Disease Control and Prevention, 2020).
Patients with PLD/CLD encounter many barriers to accessing care, including structural barriers created by insurers and medical boards. Some cannot find local clinicians to treat them, and many report that their care is not covered by insurance (Johnson, 2019a, 2019b; Johnson et al., 2011; Johnson et al., 2020; Johnson et al., 2014). These barriers to care for PLD/CLD patients were discussed extensively in the 2020 Tick-Borne Disease Working Group Report to Congress and are briefly summarized below:
- 50% of participants in the MyLymeData patient registry report that their clinicians do not accept insurance coverage;
- 26% report that they cannot find a clinician who treats PLD;
- 18 % report that they do not use antibiotics because their insurance will not cover them (Johnson, 2019a)
- 67% report that they have postponed or avoided medical treatment because of discrimination, disrespect, or difficulty obtaining care, and nearly half (47 percent) report that they have been denied treatment (Johnson, 2019a);
- 60% of survey respondents report traveling more than 50 miles for Lyme disease treatment, with 9% traveling more than 500 miles (Johnson et al., 2011);
- 82% of PLD/CLD patients who sought care at their local hospital had difficulty obtaining treatment (Johnson et al., 2011);
- 78% of patients with PLD/CLD experienced a diagnostic delay of six months or more (Johnson et al., 2014); and
- 51% saw seven or more clinicians before being diagnosed (Johnson et al., 2011).
Patient Barriers in Select Subpopulations of Patients with Tick-Borne Diseases
The mistaken practice of applying Lyme disease surveillance case criteria as diagnostic criteria may be a barrier that prevents certain patient populations from being accurately and promptly diagnosed and treated (Bacon et al., 2003; Dressler et al., 1993; Hilton et al., 1996; Sivak et al., 1996). Surveillance case definitions are epidemiologic tools that standardize data collection in order to identify risk and track trends at a population level. Disease tracking necessitates the use of narrow inclusion criteria to avoid including “non-disease” cases, which could skew the data and result in faulty conclusions. Therefore, surveillance case criteria are not intended to identify all actual cases of an illness (Mead, 2004). With regard to Lyme disease, the erythema migrans (EM) rash is the most recognizable sign of the infection; yet, as discussed below, it may be an unreliable marker in some patient populations. The 5 cm size cut-off for the rash required by the surveillance case definition may exclude others. Similarly, the serologic criteria selected for the surveillance case definition may not be clinically appropriate for all patients.
Application of the Lyme disease surveillance case criteria to clinical research has important implications. Although use of these criteria produces a homogeneous group with a high likelihood of having Lyme disease, such use excludes a significant segment of the larger Lyme disease population from being studied.
Taken together, policies and processes that promote the use of Lyme disease surveillance case criteria as diagnostic and entrance criteria for research studies are structural determinants of health that perpetuate the health disparities experienced by many patients with Lyme disease.
Blacks and Hispanics: Health disparities related to TBDs exist along racial and ethnic lines. One frequently cited study found that in a high-incidence area for Lyme disease, the incidence of Lyme arthritis was higher in Blacks than Whites, while Whites had higher rates of EM (Fix et al., 2000). A 2019 case report detailed a 32-year-old Black man whose Lyme disease was misdiagnosed despite his recent camping trip, history of a potential “bug bite,” and the presence of “an oval, red skin lesion on his posterior proximal arm, chills, and fatigue” (Dennison et al., 2019). The authors of these two papers hypothesized that under-recognition of EM in people of color may contribute to diagnostic delays (Dennison et al., 2019; Fix et al., 2000). The lack of pictures depicting EM in people of color supports that hypothesis (Bax et al., 2021; Dennison et al., 2019; Fix et al., 2000), as does a medical student’s report on the limited instruction she received about the appearance of EM rashes on people of color (Nolen, 2020). Limited access to care and implicit bias may also contribute to the diagnostic delays experienced by Blacks (Dennison et al., 2019; Fix et al., 2000; Hall et al., 2015).
Hispanics also experience disparities. Like Blacks, Hispanics are more likely than non-Hispanics to present with manifestations of disseminated disease and less likely to have a reported EM (Nelson et al., 2016). These inequities may be a function of differences in Lyme disease awareness, language barriers, and limited access to health care (Nelson et al., 2016); a survey of Hispanics and non-Hispanics in Maryland and Virginia found that Hispanics were less familiar with the vector and symptoms of Lyme disease (Hu et al., 2019). This finding has important implications regarding the risk of contracting Lyme disease because many Hispanics are employed in landscaping-related services, which is a high-risk occupation (Nelson et al., 2016).
Sex: Although few studies have examined the potential sex-based differences among patients with Lyme disease, various aspects of Lyme disease may affect males and females unequally. Studies on both animal models and humans have shown that sex-specific factors influence both the host immune response to infection and the clinical presentation of diseases caused by infectious agents (Klein & Flanagan, 2016; van Lunzen & Altfeld, 2014). For Lyme disease, these factors might increase the disease burden on women, particularly with respect to developing post-treatment Lyme disease (PTLD) (Aucott et al., 2022).
Sex-based differences in clinical aspects of early, late, and post-treatment Lyme disease have been demonstrated (Aucott et al., 2022; Rebman et al., 2021; Steere & Angelis, 2006), yet the reasons for these differences are unknown and merit further study.
A prospective study of adult patients with EM lesions measuring 5 cm or greater found that the average EM size was 2.18 cm larger in males than in females (Rebman et al., 2021). Because the CDC surveillance case definition for Lyme disease requires that an EM measure 5 cm or greater to be a reportable case, it is unclear whether sex-related differences in EM size prevent women from meeting that criterion and being included in surveillance case reports. For the same reason, a Lyme disease diagnosis in women may be delayed or precluded by clinicians who apply the EM size criterion in their clinical practice.
Men are also more likely to be diagnosed with Lyme arthritis and neuroborreliosis, which are manifestations associated with objective clinical signs (Steere & Angelis, 2006; Strle et al., 2013). Because both genders are equally represented in early stages of acute infection, sex-based differences in later disease are not readily explained (Rebman et al., 2015b).
Women appear to have a greater risk for developing PTLD (Aucott et al., 2022). PTLD is a research definition that represents a subset of PLD/CLD patients who meet specific symptom and quality-of-life criteria. A recent longitudinal study of 234 patients who were diagnosed and treated early in their infection found that being female was a significant predictor of PTLD; women were 4.2 times more likely than males to meet PTLD rather than return-to-health criteria (Aucott et al., 2022).
The effect of gender on serologic testing is unclear. While a 2008 study found no gender-based differences (Wormser et al., 2008), a 2010 study found that among people with a positive ELISA, median ELISA values and the number of IgG bands were higher in men than women (Schwarzwalder et al., 2010). Further, a 2014 study reported a borderline difference in seroconversion, with males being more likely to be seropositive following antibiotic treatment (Rebman et al., 2015a). Many clinicians rely on serologic results to establish a diagnosis (Lantos et al., 2021). Hence, the potential for sex-based differences in serologic testing to delay or preclude diagnosis in women merits further study.
Pregnancy is an experience unique to females, and the effects of TBDs on pregnant individuals and their offspring are under-investigated. However, one study found that EM rashes in pregnant women were less likely to have the classic “bull’s-eye” appearance compared to the EM rashes of nonpregnant women (Maraspin et al., 2020). Additionally, pregnant women with early Lyme disease were less likely to present with constitutional symptoms than nonpregnant women (Maraspin et al., 2020). These diminished symptoms/findings could place pregnant women at a disadvantage for being diagnosed early in their infections, when treatment is most likely to be successful.
Maternal-fetal transmission of Borrelia burgdorferi, which may result in adverse pregnancy outcomes including fetal death, premature birth, and fetal abnormalities, has been documented (MacDonald et al., 1987; Schlesinger et al., 1985; Weber et al., 1988). The issue of whether a congenital syndrome exists for Lyme disease—as it does for syphilis and other infectious diseases—is not settled and requires careful study. To date, the few studies and reviews that have been conducted on pregnancy outcomes for women with gestational Lyme disease are limited by insufficient follow-up of the offspring (Gardner, 2001; Lakos & Solymosi, 2010; Maraspin et al., 2020; Waddell et al., 2018; Walsh et al., 2007). Maternal-fetal transmission of Babesia has also been documented and requires further study (Iyer & Goodman, 2019).
Socioeconomic groups: Health disparities in TBD due to socioeconomic status is an important consideration (Moon et al., 2021; Springer & Johnson, 2018). The risk of disseminated Lyme disease is higher in patients with a low socioeconomic status and less access to health care (Moon et al., 2021). The effect of socioeconomic status varies by TBD—data analysis from 2,695 U.S. counties and the District of Columbia found that “the incidence of Lyme disease was highest in counties with relatively higher proportions of white and more educated persons and lower poverty and crime rates; the incidence of human monocytic ehrlichiosis was highest in counties with relatively higher proportions of white and less educated persons, higher unemployment rates and lower crime rates” (Springer & Johnson, 2018).
For more information on patient barriers to care, see the 2020 Training, Education, Access to Care, and Reimbursement Subcommittee Report to the Tick-Borne Disease Working Group, “Challenges to Reducing Patient-Encountered Barriers to Achieving Health Equity for Patients with Tick-Borne Diseases” (Tick-Borne Disease Working Group, 2020b).
Structural Barriers
Structural barriers include policies and processes that exclude patients from meaningful participation in decisions that ultimately affect their ability to access quality care. Funding for scientific research and the development of clinician educational content are two activities that often exclude patients and their advocates from participating. In the case of research funding, award determination committees that lack meaningful patient representation are less robust and may not recognize the importance of research avenues that directly address the lived experiences and priorities of patients (Bendiscioli, 2019). Additionally, the processes that funding organizations follow may divert funding away from novel/innovative approaches and topics that patients value (Bendiscioli, 2019). Scoring systems that award points based on researcher reputation and track record are “research-centric”; those that award points to research identified by patients as important to improving the quality of their lives are “patient- centric.” The incentives (financial and otherwise) that drive different stakeholders in health care frequently do not align (Johnson & Smalley, 2019). Because patients are the ultimate end user of healthcare research findings and the most important stakeholder in health care, their engagement is critical to achieving the primary goal of healthcare research—namely “to [improve] health by providing beneficial care to patients” (Institute of Medicine, 2009).
The choice to include or exclude patients from funding decisions varies across government entities. Although both the National Institutes of Health (NIH) and the Department of Defense (DoD)’s Tick-Borne Disease Research Program (TBDRP) fund tick-borne disease research, their processes for awarding grants differ significantly. DoD’s TBDRP includes tick-borne disease advocates in both steps of its funding review process (assessment of scientific merit and programmatic review) while NIH does not (Davey, 2021; Purdue, 2021). The availability of clinicians knowledgeable about TBDs directly impacts outcomes for patients with TBDs, yet patients have been excluded from meaningfully participating in the development of clinician curriculum. According to the National Academy of Medicine (NAM), “the key goals of medical education include helping learners at all levels develop the ability to think critically and appraise the evidence for clinical decision making” (Institute of Medicine, 2009). In pursuing these goals, outcomes that patients deem important are foundational (Johnson & Smalley, 2019). Consider the following comment from a knowledgeable Lyme disease advocate regarding her interaction with CDC’s DVBD over the development of continuing medical education modules on Lyme disease.
The overarching sentiment I’m left with is that DVBD regards their relationship with the patient community as a marriage of convenience, not a partnership. Patient groups are communicated with when their audience is desired by DVBD, but our input is not sought when foundational decisions regarding educational content, decisions that ultimately impact our care, are being made.—Lyme disease advocate
Meaningful engagement of patients requires not only selecting patients qualified to represent the community, but also engaging them early enough in the process to make a difference in the process outcome or ultimate product (Johnson & Smalley, 2019). The Training, Education, Access to Care, and Reimbursement Subcommittee Report to the 2020 Tick-Borne Disease Working Group defined the qualifications required of meaningful patient representatives and the process for selecting these representatives:
- To serve as a meaningful representative, patients or advocates representing the persistent Lyme disease community should: a) have or have had persistent Lyme disease or be someone who serves or has served as a caregiver to a persistent Lyme disease patient; and b) should be an officer or director of a recognized and trusted patient advocacy organization representing patients with persistent Lyme disease, or someone vetted and approved by such a group.
- To ensure process integrity in the selection of patient representatives through open and transparent selection processes, including public calls for nominations and soliciting nominations through recognized and trusted patient advocacy groups representing patients with persistent Lyme disease.
The Tick-Borne Disease Working Group 2020 report to Congress included a recommendation that was intended to address some of the access to care problems identified and would be relatively easy to implement (Tick-Borne Disease Working Group, 2020a). Unfortunately, this recommendation has not been implemented. Recommendation 7.1, set forth below, focused on educating the public and clinicians that the scientific understanding of Lyme disease was limited and emerging, that divergent treatment approaches existed, and that shared medical decision-making had an important role in the treatment of patients with PLD/CLD:
Recommendation 7.1: Recommend Federal government websites and educational materials and seminars for clinicians, the public, and public health departments, which discuss Lyme disease, provide information that the state of the science relating to persistent symptoms associated with Lyme disease, is limited, emerging, and unsettled; and increase public awareness that there are divergent views on diagnosis and treatment. Consider that shared medical decision-making may be appropriate in some circumstances.
Opportunities for Reducing Patient-Encountered Barriers to Achieving Health Equity for Patients with Tick-Borne Diseases
- To decrease the demand for medical services by reducing the number of PLD/CLD cases. This is best accomplished by (a) preventing disease through appropriate public awareness campaigns and (b) diagnosing and effectively treating cases of Lyme disease as early as possible.
Local public health authorities and clinicians should coordinate public awareness programs for Lyme disease and other TBDs that are locale specific in terms of potential diseases and population-specific risks.
Primary prevention of TBDs includes a whole host of strategies to avoid and prevent tick bites. These strategies have been covered in past Tick-Borne Disease Working Group reports. In the case of Lyme disease, antibiotic prophylaxis of a known blacklegged tick bite is also available (Cameron et al., 2014; Lantos et al., 2021). However, because the evidence from the largest U.S. trial is of low quality, the optimum approach continues to be debated (Sutton & Spry, 2019).
Secondary prevention involves treating the disease as early as possible, utilizing antibiotic regimens that maximize the potential to cure. The optimal duration of therapy is uncertain for many presentations of Lyme disease. New PLD/CLD cases stem largely from (a) patients who are diagnosed and treated early but remain ill after standard treatment and (b) patients who are diagnosed later in the course of their disease, when conventional treatments fail more frequently (Aucott et al., 2022; Hirsch et al., 2020; Logigian et al., 1999; Shadick et al., 1994). A recent prospective study of patients with EM rashes who were promptly diagnosed and treated reported that only 57% were categorized as “returned to health” (Aucott et al., 2022). Of the remainder of patients, 29% had persisting symptoms alone and 14% had PTLD. Among those with EM rashes who were treated with 20 days of oral amoxicillin, cefuroxime, and doxycycline, some subsets of this cohort appeared to have worse outcomes than others (Maloney, 2021).
Although recent DVBD guidance for the treatment of patients with EM rashes seeks to lessen treatment durations, in part to reduce antibiotic resistance in the population at large (Centers for Disease Control and Prevention, 2021c), this approach may be counterproductive. Given the most recent study showing significant treatment failures in optimally diagnosed and treated EM patients, efforts to shorten initial antibiotic therapy may simply result in more patients remaining ill (Aucott et al., 2022). These patients may subsequently receive additional antibiotic therapy. Further, there is no evidence that reducing treatment durations from 20 days to 10-14 days will have any meaningful effect on antibiotic resistance. - To reduce disease burden in people of color by raising their awareness of Lyme disease and other TBDs through targeted outreach efforts and by educating clinicians about increased risks and disease presentations in this patient subset.
- To open new areas of research with regard to potential sex-based differences in Lyme disease and their impact on diagnosis and treatment outcomes.
- To promote patient-centered research and medical care by including patients in research funding processes and clinician education.
Clinician-Encountered Barriers to Improving Health Equity for Patients with Tick-Borne Diseases
The supply and demand imbalance for patients with late presentations and PLD/CLD has been evaluated from the patient (Johnson et al., 2011) but not the clinician perspective. The clinicians caring for patients with complex cases of Lyme disease and PLD/CLD often navigate a variety of obstacles (Johnson, 2022). As discussed below, the lack of treating clinicians is directly related to the structural and societal barriers that complicate caring for this marginalized patient group. Between September 23 and December 1, 2021, LymeDisease.org conducted a survey of U.S. clinicians who treat PLD/CLD patients, with the objective of characterizing the types of clinicians that provide care and the barriers they encounter. The organization distributed the survey through a variety of methods including its physician referral program and broader email outreach; 155 clinicians from 30 states responded.
Most respondents (81%) treat a variety of conditions, including Lyme disease. Lyme disease dominates many practices; 57% devote more than one-half of their practice to Lyme disease. Respondents have extensive experience treating PLD/CLD patients—54% primarily treat PLD/CLD, and 57% have treated more than 500 patients with Lyme disease.
Fundamentally, the survey responses paint a picture of structural barriers that are difficult for clinicians to surmount and that disincentivize other clinicians from treating patients with Lyme disease and/or TBD. This has created a shortage of clinicians who provide care for patients with PLD/CLD and has increased the economic challenges faced by clinicians in providing care and by patients in receiving care. Although many of the individual barriers noted by the clinicians are not unique to Lyme disease (Brown et al., 2002; Friedberg et al., 2014), the accumulated obstacles, and the comments from survey participants, highlight the issues that ultimately impact access to care. Clinicians responding to the survey identified the following challenges to providing care:
- Complexity of care (79%)
- Patients’ inability to pay out-of-pocket costs (75%)
- Lack of professional support from colleagues (61%)
- Opposition to the treatment of PLD/CLD from some physician organizations (59%)
- Cognitive impairment of patients (57%)
- Frequent patient calls between scheduled appointments (49%)
- Reimbursement for care (34%)
- Length of visits (31%)
- Communication issues with patients (22%)
Challenges to Reducing Clinician Barriers and Improving Health Equity for Patients with Tick- Borne Diseases
Scientific Knowledge Gaps
“Lyme is a disease that we are just scratching the surface on.”—clinician survey respondent
The gaps in scientific knowledge of tick-borne diseases create uncertainty for clinicians who provide PLD/CLD care. Seventy-two percent of survey respondents believed that diagnostic and therapeutic uncertainty are major challenges that prevent other clinicians from treating patients with PLD/CLD (Johnson, 2022).
“Lyme disease is the hardest diagnosis I treat …; it is hard to know where to start with each one [patient], what will work, what will make them worse, etc.” – clinician survey respondent
The knowledge gaps noted by clinician survey respondents are substantial and acknowledged by others (Bobe et al., 2021; Donta, 2021; Tick-Borne Disease Working Group, 2018a, 2020b). Chief among these gaps are:
- Disease pathogenesis of the individual infections and concurrent tick-borne illnesses as well as conditions such as alpha gal syndrome,
- The need for accurate diagnostic testing, especially for patients with PLD/CLD, and
- The need to determine the optimal treatment approach for patients with TBDs, particularly PLD/CLD.
Table 1 lists the scientific gaps identified in the 2018 and 2020 Tick-Borne Disease Working Group reports (Tick-Borne Disease Working Group, 2018b, 2020a).
| Topic | Scientific Knowledge Gap |
|---|---|
| Disease surveillance |
|
| Diagnostic testing—Lyme disease |
|
| Testing for other TBDs |
|
| Ecology of tick-borne pathogens |
|
| Tick and tick-host interactions |
|
| B. burgdorferi transmission |
|
| Tick-borne pathogens-host interactions |
|
| Lyme disease pathophysiology |
|
| Persistent Lyme disease/chronic Lyme disease |
|
| Lyme disease treatment |
|
| Other TBDs and co-infections |
|
| Alpha gal syndrome |
|
Professional Stigma
“While my patients are generally supportive, some of my colleagues have stopped speaking to me and I worry about the medico-legal repercussions of what I do.”—clinician survey respondent
“I am anxious about being identified and called out by my colleagues but I feel I must treat patients because so many suffer and cannot find the care they need. I started treating about a year ago, before that I was just diagnosing and referring out. Now Cleveland Clinic refers to me. They diagnose but decline to treat!”—clinician survey respondent
Clinicians responding to the survey noted a lack of support from colleagues and professional stigma as important barriers to providing care. Seventy-five percent reported having been stigmatized or treated disrespectfully by professional colleagues because they treat Lyme disease. Additionally, 85% stated that professional marginalization is preventing other clinicians from caring for patients with PLD/CLD. One expert explains the issue of stigma and its impact in health care clearly: “Stigma is a powerful social process that is characterized by labeling, stereotyping, and separation, leading to status loss and discrimination, all occurring in the context of power” (Nyblade et al., 2019). Stigmatization and discrimination may be institutionalized in policies, procedures, or practices (such as guidelines). Clinician marginalization reduces opportunities for sharing on-call and hospital responsibilities and stifles information sharing. Exclusion from insurance networks, limited opportunities to share office space, and disparagement by colleagues are examples of stigmatization. The following excerpt describes one clinician’s experience with an insurer; see Appendix 2 for the complete statement.
“The first indication of trouble arrived unexpectedly, by registered letter. Reading in disbelief, I learned that an insurance company’s credentialing committee had reviewed a quality of care case concerning my treatment of Lyme disease and, without ever speaking to me, determined that I ‘provided inappropriate diagnosis and treatment of tick-borne illnesses.’ The letter went on to inform me that I was terminated from the insurer’s provider network. No details regarding the care concerns were provided.
Exclusion for the network would be a death blow to my practice, which provided the full range of primary care services in a small community where this particular insurer dominated the market.…And so, I was forced to pick between the small group of marginalized, suffering Lyme disease patients that I had helped and my non-Lyme disease patients who made up the bulk of the practice. With a heavy heart, I chose the latter.”—Clinician 1
Professional Risks from Regulatory Actions
“I used to practice in a state where physicians who treat complex patients including people with chronic Lyme, were specifically targeted by health insurance companies for medical board
complaints and other attacks… Eventually I elected to move to [a state] where there is less interruption of care and more protection of vulnerable patients from predatory insurance entities.”—clinician survey respondent
As reflected in survey comments, professional risks posed by regulatory bodies and insurers are a concern for many clinicians. Thirty-nine percent of respondents have been either reported to a medical board or insurer, or subjected to a hospital-based quality improvement inquiry. Clinicians reported that the potential for investigation by regulatory boards is wearing and that defending oneself requires valuable time, energy, and funds (even when sanctions are not imposed). Further, investigations are often drawn out, sometimes for years, and when investigations intrude on clinic operations, clinicians may suffer revenue losses. The following excerpts highlight the challenges that clinicians face when interacting with licensing boards (see Appendix 2 for the complete clinician statements).
“From 2003 to 2006, at the instigation of the state Board of Medical Examiners (BME), I endured a drawn-out investigation by the state Board of Nurse Examiners (BNE). Without warning, officers of the BNE arrived at my office and demanded that I immediately turn over multiple documents; not an easy task in the midst of a busy clinic day. I felt like a criminal who was being raided by the FBI. I was forced to hire an attorney who viewed the mounting allegations as harassment. Ironically, during this 3-year ordeal I was honored as my state’s Nurse Practitioner of the Year.
The BNE’s investigation produced no significant findings and I paid only a $200 fine. However, attorney fees and the intense stress of the experience left me scarred.”—Clinician 2
“I was initially the subject of a Utilization Review inquiry by a regional health carrier in 2000.
… I wrote three referenced responses to their inquiry and the investigation was discontinued.…After receiving two additional cases, the medical board reopened the investigation in 2011. Every case had been diagnosed and treated for Lyme disease. None of the patients were identified because of an adverse event, poor outcome, or malpractice. The investigator focused on the physical examination and differential diagnosis.
The prosecutor verbally offered my attorney a six-month suspension of my license followed by probation for a period of 2.5 years under specified terms and conditions for professional misconduct. Because the attorney was unable to negotiate better terms with the prosecutor, I asked the courts for a dismissal of the charges. Despite accruing more than $500,000 in legal fees, I lost the appeal.”—Clinician 3
State medical boards, hospital credentialing committees, and insurer quality committees may be ill- equipped to judge care when a medical field is evolving and relevant trial evidence is scarce. Although many of these bodies rely on their perception of the “standard of care” and prevailing clinical practice guidelines (CPG), these metrics are of limited use in Lyme disease, where the evidence base is weak and treatment approaches diverge (Johnson et al., 2018).
Standard of care for Lyme disease can be difficult to define. Common definitions of medical standard of care refer to the care that a reasonably prudent and skilled healthcare professional with similar training practicing in the same community would provide under a given set of circumstances. With regard to Lyme disease, the applicable standard of care may depend on what disease stage the clinician is treating. Because few patients with PLD/CLD are treated by infectious disease specialists (Johnson et al., 2011), it seems logical to base the PLD/CLD standard of care on the experiences of the clinicians who treat patients with PLD/CLD rather than solely on the experiences of infectious disease specialists.
As described above, PLD/CLD is a research-disadvantaged disease, similar to rare diseases, with few incentives driving treatment research. Treatment for PLD/CLD typically consists of generic off-label antibiotics. Clinical innovation and off-label treatments are often seen in diseases (for example, cancer) for which clinical needs exceed the available evidence base (Tabarrok, 2000). Given the heterogeneity of the PLD/CLD population and its response to treatment (Johnson et al., 2018), individualized care is customary. Hence, no single uniform course of treatment for this population is recognized as desirable, and the need for clinical innovation to address the needs of patients with PLD/CLD is strong. However, successfully innovative clinicians treating PLD/CLD have found themselves sanctioned for being ahead of their colleagues. The same logic applies to the use of CPGs. Guidelines adopted/endorsed for the treatment of patients with PLD/CLD should include the expertise of the clinicians who actually provide this care.
Some regulatory decisions that sanction clinicians who treat patients with TBDs may reflect misconceptions regarding the available evidence, misunderstandings regarding the principle and practice of evidence-based medicine, and misgivings or a lack of appreciation of shared decision-making. In addition to the significant scientific gaps listed in Table 1, the 2018 and 2020 Tick-Borne Disease Working Group reports identified several clinician knowledge gaps (see Table 3), which include:
- Insufficient understanding of the spectrum of clinical presentations of TBDs;
- Underestimation of their patients’ risk of Lyme disease;
- Mistaken belief that patients who do not meet the case definition cannot have Lyme disease;
- Insufficient education regarding the limitations of testing in tick-borne illnesses, including the potential for false positives and false negatives; and
- Lack of understanding regarding treatment (Tick-Borne Disease Working Group, 2018a, 2020b).
Regulators may enlist outside experts to assist in investigations related to the diagnosis and treatment of TBDs (Martinez, 2021). However, when the experts themselves are unfamiliar with the pertinent research evidence and harbor some or all of the knowledge gaps listed above, it increases the likelihood that they will primarily support their opinions by relying on popular third-party sources such as CPGs. The inherent problem with this approach is that CPG recommendations often provide a generalized, population-based approach to patient management that cannot account for the details of an individual patient’s circumstances, treatment needs, or values and preferences (Djulbegovic & Guyatt, 2017). Therefore, when the patient population is heterogeneous and patients require individualized care, regulatory decisions based chiefly on CPG recommendations reflect a flawed process that is unfair to the clinician under investigation.
Clinicians and regulators aim to consistently apply evidence-based medicine in the clinical management of patients, yet many clinicians, including those who serve on regulatory bodies, may have a fundamental misunderstanding of evidence-based medicine. As defined by its founders, evidence-based medicine “is the integration of best research evidence with clinical expertise, and patient values” (Sackett et al., 2000). These elements exist in tension with one another, and the value of each is situational. When the clinical research evidence is strong, it is weighted more heavily than clinical expertise and patient values. This is because clinical expertise and patient values would likely align with the research findings. In contrast, clinical expertise may diverge from the trial findings when the research evidence is weak. In such circumstances, the spectrum of patient values may be quite broad. Thus, when the evidence is weak, it is afforded less weight while the importance of clinical expertise and patient values rises.
Although several randomized clinical trials have been conducted for Lyme disease, the quality of the evidence continues to be debated (Cadavid et al., 2016; Cameron et al., 2014; Lantos et al., 2021; National Institute for Health and Care Excellence, 2018); two of the three organizations reviewing the evidence base generally rated the quality as “low” (Cameron et al., 2014; National Institute for Health and Care Excellence, 2018). One central concern with the evidence base is that the treatment trials were small, used average treatment effects, and excluded the majority of patients seen in clinical practice.
Therefore, applying the results from these trials to the heterogeneous Lyme disease patient population that most clinicians encounter is problematic. Although clinicians treating patients with TBDs, especially those who treat patients with PLD/CLD, may be aware that the clinical research with regard to therapeutic benefit is not of high quality, those who are evaluating their care may not. Evidence quality is directly linked to the strength of a treatment recommendation; strong recommendations require strong evidence, while weak evidence begets weak recommendations. As Dr. Gordon Guyatt, who played a key role in the development of GRADE (Grading of Recommendations, Assessment, Development, and Evaluations) explained in his presentation to this Subcommittee:
“Weak recommendations are [patient]value and preference sensitive.…A strong recommendation is a candidate for a quality assurance criterion, a weak recommendation is not because the right thing to do differs from patient to patient. The only quality criterion for a weak recommendation is that physicians make an effort to ascertain patients’ values and preferences in making a decision” (Guyatt, 2021).
Evidence-based medical care should also be patient-centered. Shared decision-making is the process through which clinicians come to understand the goals, values, and preferences of their patients while informing the patients of their options and the risks and benefits associated with each option. In discussing uncertainty in medicine with this subcommittee, Dr. Richard Schwartzstein explained that in the face of uncertainty, “High value care is largely making the care consistent with the values of the patient” (Schwartzstein, 2021).
Traditional Economic Models Ill-Suited to the Care of PLD/CLD Patients
Survey responses indicate that the traditional medical economic model does not work in the context of PLD/CLD (Johnson, 2022). The traditional model relies heavily on insurance compensation, which necessitates that clinicians be an in-network provider who primarily offers brief patient appointments. Yet, 70% of respondents reported that initial consults last 1-2 hours or more (25% > 2 hours; 44% - 1 to 2 hours). Longer follow-up visits are common as well, with 40% of respondents reporting durations of 1 or more hours. Because insurance compensation often does not match the time required to provide care, 74% of respondents do not participate in insurance networks and 77% do not participate in Medicare, Medicaid, or other government-supported plans. Therefore, many clinicians (74%) provide care on a cash-only basis. Yet, 75% of respondents identified the inability of patients to pay out-of- pocket expenses as a major challenge to providing care.
Need for Increased Research Funding
Research funding is critical to reducing the numerous gaps in the scientific evidence related to TBDs, yet NIH has historically allocated insufficient funds to investigate this group of diseases. The NIH “Table of Estimates of Funding for Various Research, Condition, and Disease Categories (RCDC),” published on June 25, 2021, lists funding details for 2020 across 299 research areas (https://report.nih.gov/funding/categorical-spending#/). A similar table for 2021 is not yet available.
Table 2 compares NIH funding for Lyme disease and tuberculosis (TB). To compare spending per case, 2019 data were used because the pandemic may have artificially lowered the reported incidence of TB in 2020 (Deutsch-Feldman et al., 2021). In the United States, the annual incidence of Lyme disease is 476,000 cases and the 2019 incidence of tuberculosis was 8,910 cases (Deutsch-Feldman et al., 2021; Kugeler et al., 2021). Thus, the per case spending is $67 for Lyme disease and $54,800 for TB. This comparison is not intended to be a critique of TB funding; rather, its purpose is to highlight the need for increased funding for Lyme disease, including research involving Lyme disease patient subgroups that to date have been underrepresented (Bernard et al., 2021).
| Disease | 2016 | 2017 | 2018 | 2019 | 2020 | 2021* | 2022* |
|---|---|---|---|---|---|---|---|
| Lyme | $28 | $22 | $30 | $32 | $40 | $50 | $50 |
| Tuberculosis | $290 | $347 | $403 | $488 | $477 | $493 | $503 |
Dollars in millions; *estimated
The $40 million in funding for Lyme disease in 2020 was distributed across 92 projects. The NIH table of funded projects may include clerical errors because it indicates that slightly more than $3 million was directed toward lupus and tick-borne flavivirus infections. Other government programs also fund Lyme disease research on a smaller scale; the Congressionally Directed Medical Research Program (CDMRP) awarded $7 million to the Tickborne Disease Research Program (TBDRP) in both 2020 and 2021.
Opportunities to Reduce Clinician Barriers and Improve Health Equity for Patients with Tick-Borne Diseases
- To reduce stigma and professional risks faced by clinicians who treat PLD/CLD and other complex cases of TBDs by educating all clinicians and regulatory bodies about the uncertainty surrounding the diagnosis and treatment of these illnesses. This education should also address misconceptions and misapplications of clinical practice guidelines, evidence-based medicine, and shared decision- making. Such actions would likely lead to an increase in the number of clinicians willing to care for this group of marginalized patients.
- To develop innovative research methods, including the development and growth of community research capacity to accelerate the fundamental knowledge base using “big data” registries and databases, data-sharing platforms, specimen and tissue sample repositories, as well as genomic and precision medicine approaches that reflect the underlying heterogeneous nature of PLD/CLD. This effort may include leveraging existing NIH resources and expanding collaborations across NIH Institutes to promote a multidisciplinary approach to TBD research with, for example, the NIH Community-Based Participatory Research Program (https://www.nimhd.nih.gov/programs/extramural/community-based-participatory.html). The ethical rationale and principles for patient engagement in research were established in community-based participatory research (CBPR). CBPR provides a framework to respond to health issues within a social and historical context while reducing mistrust of the people being studied, through collaboration and partnership between the researcher and community. The ultimate goal is for the community to own the results and use them to improve health outcomes and quality of life (Johnson & Smalley, 2019; Macaulay, 2017).
- To promote ongoing research to investigate potential mechanisms of B. burgdorferi persistence that may influence therapeutic choices. Studies on persister (antibiotic tolerant) B. burgdorferi organisms and the bacteria’s response to antibiotic stress in regard to their potential relationship with the symptoms and signs experienced by PLD/CLD patients could prove to be clinically useful (Feng et al., 2019; Sharma et al., 2015). Some investigators have raised concerns regarding the existence and potential role of borrelial biofilms (Gindl et al., In review). Research directed to this topic may also be clinically relevant.
- To develop novel therapeutics for PLD/CLD and other TBDs, such as babesiosis, that are seeing a diminished effectiveness of prevailing treatment regimens (Renard & Ben Mamoun, 2021). In addition to newly developed drugs, this work might investigate the therapeutic potential of novel combinations of approved drugs and repurposed medications.
- To develop solutions to the failed economic care delivery model in PLD/CLD that precludes most clinicians from participating in insurance networks, threatens clinical practice sustainability, and renders many patients unable to pay for care.
Medical Education-Related Barriers to Improving Health Equity for Patients with Tick-Borne Diseases
Improving health equity for patients with TBDs requires a sufficient number of well-educated clinicians whom patients can readily access. Both the 2018 and 2020 Tick-Borne Disease Working Group reports identified clinician knowledge gaps that impact the diagnosis and management of patients with tickborne illnesses (see Table 3 for details)(Tick-Borne Disease Working Group, 2018a, 2020b). The 2020 Tick-Borne Disease Working Group report included four recommendations regarding clinician education; Recommendation 7.2, which specifically focused on Lyme disease education, underscored the need to “increase the pool of qualified and practicing clinicians”(Tick-Borne Disease Working Group, 2020a). The continued need for clinician education that promotes an increased awareness of TBDs and fosters a deeper understanding of these illnesses remains a significant concern of this subcommittee.
| Topic | Clinician Knowledge Gap |
|---|---|
| General |
|
| Rickettsial, ehrlichial, and anaplasmal diseases |
|
| Lyme disease |
|
| Babesiosis |
|
| Alpha gal syndrome |
|
| Sources: (Tick-Borne Disease Working Group, 2018b, 2020a) | |
Challenges to Reducing Medical Education-related Barriers to Improving Health Equity for Patients with Tick-Borne Diseases
Uncertainty
Uncertainty in the TBD field produces clinical uncertainty. As the scientific knowledge of TBDs continues to grow and evolve, many uncertainties remain or are just coming to light. For example, the discovery of new pathogenic Borrelia species, especially B. miyamotoi, whose distributions and symptoms overlap with those of other TBDs, introduces diagnostic uncertainty—does this patient have Lyme disease or B. miyamotoi disease? The expansion of tick ranges poses similar problems. The lone star tick (Ambylomma americanum), which is associated with southern tick-associated rash illness (STARI), is now widely distributed in the eastern United States (Kennedy et al., 2021). Although the initial presentation of the illness is similar to acute Lyme disease, the pathologic agent of STARI has yet to be identified and no diagnostic tests for this infection exist. Thus, although the EM rash used to be considered pathognomonic for Lyme disease, when patients present with an EM rash, clinicians in parts of the country must also consider STARI as a potential diagnosis.
Diagnostic uncertainty has two main sources. Uncertainty related to clinical presentations is one. This includes the challenges posed by the overlapping clinical presentations of many TBDs and the highly variable presentation of disseminated Lyme disease. The limitations of diagnostic testing is the other. Point-of-care testing has limited availability, and many diagnoses are based on serologic tests, which can be inaccurate for many reasons (Bobe et al., 2021), including that antibody responses in a specific individual are unpredictable and may not fall within established laboratory norms (Aguero-Rosenfeld et al., 1996; Steere et al., 2008). With regard to PLD/CLD, no tests exist to demonstrate successful bacterial eradication or to confirm other potential etiologies for ongoing symptoms and signs (Centers for Disease Control and Prevention, 2021a).
Therapeutic uncertainty remains a concern. For many TBDs, prevailing therapeutic regimens are based on clinical observations with little or no evidence from randomized controlled/comparative trials.
Although the body of evidence for Lyme disease is greater than those for other TBDs, it is still quite limited and the quality of that evidence is the source of disagreement (Cadavid et al., 2016; Cameron et al., 2014; Hayes & Mead, 2004; Lantos et al., 2021; National Institute for Health and Care Excellence, 2018); the majority of GRADE- based evidence assessments regarding treatment benefits determined that the evidence is generally of low quality (Cadavid et al., 2016; Cameron et al., 2014; Hayes & Mead, 2004; National Institute for Health and Care Excellence, 2018).
Rapidly Expanding Medical Knowledge
The dramatic increase in medical knowledge presents an important challenge in TBDs. Medical knowledge, as measured by journal articles and other data metrics, has been increasing exponentially for over a decade and is only likely to increase faster in decades to come. In contrast, instructional time in undergraduate medical education is essentially fixed. Thus, the need to cover more information in a fixed amount of instructional time results in educational trade-offs. As such, TBDs compete with other high-impact, high-incidence topics such as diabetes and obesity for learning time. Viewed in that light, it is understandable that limited classroom time is spent on TBDs.
The implications of this educational choice profoundly affect patients with TBDs. Clinicians generally leave school with limited knowledge of basic facts of this group of illnesses and with little exposure to the uncertainties mentioned above. Therefore, clinicians often struggle to (a) identify many disease presentations, (b) determine how to assess an individual’s risk of disease based on history and exam, (c) recognize the limits of testing, and (d) individualize care. These struggles are reflected in the lived experiences of many patients who found it difficult to obtain a diagnosis and treatment when they first sought medical care (Tick-Borne Disease Working Group, 2018b).
Increasing Need for Self-Directed Learning
As the complexity of TBDs becomes better known, clinicians must be able to incorporate additional information into their practice. Thus, clinicians must be life-long learners (Ginzburg et al., 2021). Although self-directed learning is beneficial in medical training (Murad et al., 2010) as well as in post- graduate and continuing education, it is not clear that physicians leave formal medical training with this skill (Premkumar et al., 2013). Self-directed learning requires individuals to identify their learning needs, set goals, identify resources for learning, and implement a strategy to meet the goals. This process requires time, opportunities, and available resources. Continuing medical education (CME) opportunities are part of the resources on which clinicians rely to remain life-long learners.
The characteristics of CME opportunities evaluated by healthcare professionals in determining which courses to pursue are cost, ease of access, perceived provider reliability, and flexibility, that is, convenient for the learner’s schedule (O'Brien Pott et al., 2021a, 2021b). Clinicians report that expense and travel time are their largest barriers to obtaining CME. The most frequently used sources of CME course information are online searches and e-mail (O'Brien Pott et al., 2021a). As part of this subcommittee’s assessment of the state of TBD education, an informal Google search looking for online CME for Lyme disease was conducted by one member (BS). The search results were assessed based on the above qualities. Few unique online, low or no-cost, flexible CME opportunities were identified; included among those were the CDC TRAIN Clinician Education Modules pertaining to Lyme disease.
CDC TRAIN Continuing Medical Education Modules on Lyme Disease
Given the approximate 500,000 cases of Lyme disease occurring annually in the United States and prior calls to improve clinician education, it is natural that the CDC would take a lead role in providing comprehensive, factual, up-to-date information on Lyme disease that clinicians could rely on. CDC self identifies, and is regarded by others, including the public, healthcare providers, insurers (BlueCross BlueShield of North Carolina, 2021a, 2021b), and professional medical boards, as a respected and authoritative source of information regarding health topics including Lyme disease and other tickborne disorders. Yet, many people who rely on its expertise do not realize that CDC approaches individual diseases and topics from a population-based perspective (Partridge, 2021). This perspective differs from and often conflicts with the primary concerns of clinicians, who are providing patient-centered care to specific individuals. The CDC TRAIN CME pertaining to Lyme disease is a series of four, one-hour modules on different aspects of Lyme disease. It was released in August 2021 as an online CME offering. In many ways, the modules do not meet the educational needs of clinicians. This report highlights a few areas of concern:
- The content does not accurately reflect the current body of scientific knowledge. Specifically, the modules are not forthcoming about what remains unknown, what is uncertain, and what continues to be debated. Instead, the instructional materials selectively present information that paints a simplified picture of the current state of the science. This not only limits the clinical utility of the information presented but gives the impression of settled science where it does not exist. These are missed opportunities to inform clinicians about the uncertainty regarding the treatment, especially treatment of PLD/CLD.
- The modules lack references, which is unusual in accredited CME. Lacking that information, participants cannot identify the source material and thus cannot reach conclusions regarding the rigor of science being presented, as well as potential errors or biases in the presentations.
- The modules do not provide detailed information regarding potential disease symptoms and signs. Lyme disease diagnoses are often based on subtle aspects of a patient’s history and exam. In the absence of a reported known bite or obvious EM rash, clinicians must be taught how to elicit other information that makes Lyme disease more or less likely. The modules do not adequately discuss the well-known limitations of serologic testing. The absence of important clinical teaching points represents a missed opportunity.
- The recommendations regarding antibiotic prophylaxis of a known blacklegged tick bite and the other CDC treatment recommendations do not appear to reflect a systematic review of the evidence nor do they acknowledge the low quality of the treatment-related evidence identified by others (Cadavid et al., 2016; Cameron et al., 2014; Hayes & Mead, 2004; Hu & Shapiro, 2021; National Institute for Health and Care Excellence, 2018). In the case of antibiotic prophylaxis of a known tick bite, the modules fail to inform clinicians that the recommendation is based on a single human trial conducted in the U.S.
- The messaging pertaining to the success/failure of antibiotic retreatment in Lyme disease does not accurately portray the findings of the underlying studies. Successful antibiotic retreatment following initial treatment failure was demonstrated in several U.S. trials, but these findings are not noted in the treatment module (Dattwyler et al., 1997; Eppes & Childs, 2002; Logigian et al., 1990, 1999; Luft et al., 1996; Luger et al., 1995; Massarotti et al., 1992; Nadelman et al., 1992; Wormser et al., 2003).
- The discussion of the four-NIH sponsored trials of antibiotic retreatment of persistent manifestations of Lyme disease following initial antibiotic therapy is inaccurate. Two of the four trials demonstrated a sustained beneficial treatment effect in the subset of patients with severe fatigue (Fallon et al., 2008; Krupp et al., 2003). Although discussions in the scientific literature regarding the findings from the retreatment trials are quite nuanced and remain subject to debate (Delong et al., 2012; Fallon et al., 2012; Klempner et al., 2013), ignoring the discussion altogether in favor of a generalized retreatment doesn’t work posture misleads clinicians on a topic that is critical to the care of patients with PLD/CLD.
The process that CDC’s DVBD followed to develop the CME modules is not entirely clear. However, it appears that it did not follow the process for developing treatment recommendations that is laid out in the 2002 HHS publication Guidelines for Ensuring and Maximizing the Quality, Objectivity, Utility, and Integrity of Information Disseminated to the Public (Office of the Assistant Secretary for Planning and Evaluation, 2002). Those guidelines call for inclusion of stakeholders, such as patients, patient advocacy leaders, and broadly experienced clinician experts, in the development of materials. Recommendations from both the 2018 and 2020 Tick-Borne Disease Working Group reports had similar language regarding the engagement of specific stakeholder groups (Tick-Borne Disease Working Group, 2018b, 2020a). The process for selecting source material to generate content is unclear. On request, this subcommittee obtained a limited list of references; review articles were a common source document. The use of review articles rather than primary literature sources may give the impression of more current or more recent trial data supporting recommendations than actually exists. It also may predispose to the perpetuation of particular points of view, or even errors.
Development of the CME modules began in September 2019, and input from different potential user groups was solicited well before release. However, there is no indication that these groups were specifically selected because of their subject matter expertise. Feedback from groups that lack subject matter expertise would not serve to confirm whether the presentations reflected the evidence base. It was not until July 2021 that a knowledgeable patient advocate and a physician subject matter expert were asked to provide feedback to the DVBD. Despite bringing important points to light, the project was too far along for their engagement to result in meaningful changes; the modules were released in August 2021.
Taken together, the timeline for the content development process highlights the importance of involving the right (knowledgeable) stakeholders at the right time—when decisions regarding educational goals and content topics, source materials, and patient needs and goals are being made. Regardless of whether the educational modules technically fall under the 2002 guidance, it seems to this subcommittee that the over-arching intent of HHS guidance is for all information distributed by umbrella agencies to be held to these standards for high quality, utility, objectivity, and integrity.
CDC Webpages on Lyme Disease
The CDC, through its webpages on TBDs, has the difficult and important task of presenting information to a wide range of users, including the public and medical professionals. It has a responsibility to provide and discuss the available evidence accurately and acknowledge gaps in the scientific understanding of TBDs. The expansive reach of the website (Partridge, 2021) is far greater than that of the CDC education modules; thus, when educational opportunities are missed or do not reflect the state of the science, the potential for harm, rather than aid, is magnified.
Because it is not feasible for this subcommittee to address every CDC TBD webpage, this report is focused on the CDC webpages (https://www.cdc.gov/lyme/index.html) devoted to Lyme disease. Many of the following points reflect the same concerns regarding content accuracy that were raised with regard to the CME modules. The content does not provide an accurate review of the current body of knowledge and fails to acknowledge what remains unknown, what is uncertain, and what continues to be debated.
- The webpages miss the opportunity to fully describe this multifaceted disease, its many and varied presentations, and the consequences of missed and delayed diagnosis and treatment. Although much of the content is factual, it lacks sufficient depth and, at times, perpetuates common misconceptions.
- The webpages on signs and symptoms do not provide detailed information regarding potential disease symptoms and signs or elements of the medical history that make Lyme disease more or less likely. Therefore, clinicians are ill-equipped to estimate patient-specific, pre-test probabilities. The section on diagnostic testing does not adequately discuss the well-known limitations of serologic testing.
- Treatment recommendations do not acknowledge the low quality of the available evidence on which they were based and leave no apparent room for clinicians to exercise clinical judgment or engage in shared decision-making.
- Although a limited list of references follows the treatment recommendations for erythema migrans (https://www.cdc.gov/lyme/treatment/index.html), Lyme carditis (https://www.cdc.gov/lyme/treatment/lymecarditis.html), neurologic Lyme disease (https://www.cdc.gov/lyme/treatment/NeurologicLyme.html), and Lyme arthritis (https://www.cdc.gov/lyme/treatment/LymeArthritis.html) , these references do not appear to represent a systematic and comprehensive review of the pertinent evidence. Hand-cultivated lists are problematic because they often reflect the anchoring and confirmation biases of those selecting the evidence to be reviewed, which may perpetuate particular points of view, or even errors.
- The content regarding PLD/CLD (https://www.cdc.gov/lyme/postlds/index.html) appears skewed in that it does not provide clinically helpful information or references regarding the condition, potential etiologies, knowns and unknowns, or the diverse and often conflicting scientific viewpoints that mark this challenging health problem. The list of references is overly weighted toward the risks associated with treatment and provides an inaccurate assessment of the potential benefits. (See the discussion on page 38 regarding the evidence pertaining to antibiotic retreatment).
- Although information about the scientific evidence may be too detailed for the general public, it could and should be included on the “health care providers” webpage (https://www.cdc.gov/lyme/healthcare/index.html). At present, this webpage offers an inaccurate portrayal of the available scientific evidence and current state of clinical uncertainties. This is best exemplified by including a video of a patient discussing his misdiagnosis of Lyme disease while omitting any discussion or videos of patients with Lyme disease whose lives were irretrievably altered by diagnostic and therapeutic delays when Lyme disease was erroneously diagnosed as something else, or the complex problem of PLD/CLD.
NIH/NIAID Webpages on Lyme Disease
The NIH/NIAID webpages regarding Lyme disease and other TBDs, consistent with the Institute’s missions, are primarily research oriented. Citing its response to the TBDWG 2018 report ’s recommendation, NIH generated a trans-agency strategic plan for TBD research and includes it on the website (Tick-Borne Disease Working Group, 2018b). Two of the findings in this report are aligned with and further the NIH Strategic Plan for Tickborne Disease Research. Similarly, in 2019, NIAID updated its 2015 Report on Current Efforts in Lyme Disease Research. NIAID, as the primary home of TBD research at NIH, is the focus of the remainder of these comments.
The NIAID’s stated goals regarding Lyme disease are “to develop better means of diagnosing, treating, and preventing” Lyme disease via a broad range of activities “designed to increase our understanding of this disease” including “high priority areas such as persistence of infection after antibiotic treatment and the development of both early- and late-stage diagnostics.” (https://www.niaid.nih.gov/diseases-conditions/addressing-lyme-disease).
Despite a high-priority designation, the webpage’s treatment of persistence of infection and PLD/CLD does not accurately represent the available science or the controversies still being debated. Without providing references, the discussion of the NIH-sponsored clinical trials on antibiotic retreatment of PLD/CLD dismisses the beneficial treatment effects identified in the Krupp and Fallon trials (Fallon et al., 2008; Krupp et al., 2003) and disregards important methodologic issues (Delong et al., 2012; Fallon et al., 2012), which were raised about the Klempner trials (Klempner et al., 2001). Rather than examining the nuances of the trials’ designs and the divergent findings, the discussion presents two erroneous conclusions: (a) the trials proved that the antibiotic regimens, IV ceftriaxone alone or in combination with oral doxycycline, were ineffective and (b) that the perceived lack of benefit from these two regimens implies that all potential antibiotic retreatment regimens would fail. Considering that these small studies excluded 89-99% of patients who sought to enroll (Fallon et al., 2008; Klempner et al., 2013; Krupp et al., 2003), suggesting that all attempts at antibiotic retreatment would be futile is a disservice to patients and clinicians. In support of its position against antibiotic retreatment in PLD/CLD, the webpage discussion turns to the efficacy findings from a more recent European study (Berende et al., 2016), but without acknowledging the full extent of that study’s limitations or important mitigating elements.
Because the NIAID discussions do not adequately address notable evidence gaps in the research regarding treatment of PLD/CLD or the continued debate surrounding the U.S. trials, they miss the opportunity to appropriately inform visitors. The CDC website links to the NIAID pages, magnifying the reach of this content. This missed opportunity impacts patients immediately by misinforming clinicians. And, by portraying this topic as settled science, it may impact the field in a more subtle and long-term way should it dissuade researchers and government and private funders from pursuing this important area of investigation.
CDC, NIH, and NIAID are leading medical authorities within the government and exert a strong influence on medical professional organizations, regulatory bodies, health insurers, researchers, medical educators, and providers. Given this broad reach, they have a particular obligation to provide accurate and unbiased evaluations of TBD research and information, including that relating to Lyme disease. In failing to do so, they ultimately contribute to the health inequities faced by patients with Lyme disease.
Opportunities for Reducing Medical Education-Related Barriers to Improving Health Equity for Patients with Tick-Borne Diseases
- To reassess existing Federal health agencies’ TBD content and messaging, beginning with but not limited to CDC webpages and current CDC TRAIN modules pertaining to the prevention, diagnosis, and treatment of Lyme disease, and NIH/NIAID webpages referring to Lyme disease, to determine whether they best reflect the current state of the science and appropriately delineate areas of scientific uncertainty. This review will be conducted by a newly formed committee under the aegis of HHS. This committee will include meaningful stakeholder engagement of subject matter experts, which could be established under the Federal Advisory Committee Act. The committee members will include (a) patients, caregivers, or patient advocates who can meaningfully represent PLD/CLD community interests, (b) clinicians (including clinicians who treat PLD/CLD and clinicians who are subject matter experts in PLD/CLD education), and (c) research scientists with expertise/experience that represents diverse scientific perspectives on the full spectrum of Lyme disease. Stakeholders will be active members of the committee, involved with selecting priority areas for review, determining important outcomes that matter to patients, conducting the review, and assisting with dissemination of results. The aim of the review is to work in a collaborative fashion to identify the scientific knowns and unknowns of Lyme disease.
As part of its work, the committee will engage in a “Red Team” exercise to determine the strengths and weaknesses of the current CDC TRAIN educational modules and webpage content. A Red Team is defined as a team that is formed with the objective of subjecting an organization’s plans, programs, ideas, and assumptions to rigorous analysis and challenge (UK Ministry of Defence, 2021). - To produce high-quality CME modules on TBD through DVBD. To accomplish this work, DVBD should follow the process described in the “Standards Required for the Development of CDC Evidence- Based Guidelines” (Carande-Kulis et al., 2022).
This effort should meaningfully engage stakeholders from the beginning of and throughout the process. Stakeholders should include (a) patients, caregivers, or patient advocates who are members of the Lyme disease communities and who can meaningfully represent their interests, (b) clinicians (including clinicians who treat PLD/CLD and clinicians who are subject matter experts in PLD/CLD education), and (c) research scientists with expertise/experience that represents diverse scientific perspectives on the full spectrum of Lyme disease. Identifying areas of uncertainty and knowledge gaps is important in developing “accurate, reliable, clear, complete, unbiased, and useful” Lyme and TBD clinician education materials (Office of the Assistant Secretary for Planning and Evaluation, 2002). For DVBD to identify areas of uncertainty in the science, to present information on conflicting strategies for addressing Lyme disease and other tickborne disorders, and to recognize the low quality of the evidence on which recommendations are based (Cameron et al., 2014; Hayes & Mead, 2004; National Institute for Health and Care Excellence, 2018) will be an important step forward. - To increase the numbers of treating clinicians by:
- Providing clinically useful educational materials that will help support more clinicians in caring for patients with Lyme disease and all TBDs. Providing content that details specific areas of scientific uncertainty and debate will enable clinicians to exercise their clinical judgment and engage in shared decision-making with patients and should reduce some of the clinician-encountered barriers discussed earlier in this report.
- Linking knowledgeable, experienced clinicians/subject matter experts with primary care providers in a telementoring relationship.
Telementoring platforms such as Project Extension for Community Healthcare Outcomes (ECHO) have demonstrated significant impact on improving access to quality care for underserved and/or stigmatized populations through the use of video-conferencing technology to leverage a single specialist to contribute to increasing primary care capacity to deliver quality care. Utilizing this hub and spoke design, specialists can develop an ongoing collaborative telementoring relationship with clinicians across the country, which includes reach-back capability to discuss complex cases. This unique capacity to develop primary care clinicians where patients live (for example, geographic areas with limited specialists) is a primary contributor to the powerful effects of telementoring. Telementoring has expanded over time to a number of Superhubs and Initiatives (for example, Diabetes and Endocrinology; Opioid and Pain Related ECHOs; Access to Specialty Care; and HIV, TB, Health Security), but to date TBDs have not been included in Project ECHO’s ongoing collaboration with HHS (for example, Assistant Secretary for Preparedness and Response, Agency for Healthcare Research and Quality, and Indian Health Service) (Agency for Healthcare Research and Quality, 2020; Project Echo, n.d.-a, n.d.-b ).
- To produce additional educational materials for clinicians to learn lifelong skills in addressing and managing uncertainty, developing differential diagnoses, interpreting evidence, and understanding the role of clinical practice guidelines, particularly when there is disagreement in those guidelines. This content should be created in accordance with standards to produce evidence-based guidelines (above). Consider partnering with the Association of American Medical Colleges (AAMC) or other undergraduate medical education-focused regulatory bodies on the development of TBD educational materials to be used in undergraduate education.
Although the accomplishment of these goals in undergraduate medical education is outside the scope of this report, it is consequential that medical educators have identified these needs (Krupat et al., 2017; Meyer et al., 2021; Mylopoulos & Regehr, 2007) and are working to address them (Richards, 2021; Schwartzstein, 2021). The incorporation of successful undergraduate educational approaches that foster these cognitive capacities and self-directed learning practices into CME efforts should be encouraged. Consulting or partnering with the American Association for Continuing Medical Education (AACME) may help to identify ways to move that goal forward.
Findings
Priority 1 Findings
Finding 1.1. Fund and support a directive for CDC to conduct a review of CDC webpages and the current CDC TRAIN CME modules pertaining to the prevention, diagnosis, and treatment of Lyme disease to determine whether they best reflect the current state of the science and appropriately delineate areas of scientific uncertainty.
This review will be conducted by a newly formed committee under the aegis of HHS. This committee will include meaningful stakeholder engagement of subject matter experts, which could be established under the Federal Advisory Committee Act. The committee members will include (a) patients, caregivers, or patient advocates who can meaningfully represent PLD/CLD community interests, (b) clinicians (including clinicians who treat PLD/CLD and clinicians who are subject matter experts in PLD/CLD education), and (c) research scientists with expertise/experience that represents diverse scientific perspectives on the full spectrum of Lyme disease. Stakeholders will be active members of the committee, involved with selecting priority areas for review, determining important outcomes that matter to patients, conducting the review, and assisting with dissemination of results. The aim of the review is to work in a collaborative fashion to identify the scientific knowns and unknowns of Lyme disease.
As part of its work, the committee will engage in a “Red Team” exercise to determine the strengths and weaknesses of the current CDC TRAIN CME modules and webpage content. A Red Team is defined as a team that is formed with the objective of subjecting an organization’s plans, programs, ideas, and assumptions to rigorous analysis and challenge (UK Ministry of Defence, 2021).
| Number in Favor | Number Opposed | Number Abstained | Number Absent |
|---|---|---|---|
| 8 | 0 | 0 | 0 |
Minority Responses
There were no minority responses.
Finding 1.2. Fund and support modification of the Federal government websites, starting with the CDC and NIH websites, as well as educational materials and seminars for clinicians, the public, and public health departments to incorporate the following language: “the state of the science relating to persistent symptoms associated with Lyme disease, is limited, emerging, and unsettled; and increase public awareness that there are divergent views on diagnosis and treatment. [Clinicians should] consider that shared medical decision-making may be appropriate in some circumstances.”
This recommendation aligns with Finding 1.1 and implements Recommendation 7.1 from the 2020 Working Group report (Tick-Borne Disease Working Group, 2020a).
| Number in Favor | Number Opposed | Number Abstained | Number Absent |
|---|---|---|---|
| 8 | 0 | 0 | 0 |
Minority Responses
There were no minority responses.
Finding 1.3. Provide the HHS Secretary with discretionary authority to maintain telehealth flexibilities independent of Public Health Emergency declaration (for example, coverage of services in the patient’s home and in any geographic area as an “originating site,” payment parity for in-person vs. telehealth services). These flexibilities will ensure that PLD/CLD patients have access to appropriately trained clinicians to bridge gaps in current PLD/CLD clinician workforce.
Additionally, include PLD/CLD patients as a unique group requiring specialized integrated care delivery similar to Long COVID and Myalgic Encephalomyelitis/Chronic Fatigue Syndrome communities.
Interdisciplinary care needs may require expanding eligible practitioners who can furnish telehealth in Medicare (for example, physical therapists, occupational therapists) outside of the already covered telehealth services for the mental health, behavioral health, and neurocognitive dimensions of PLD/CLD (Rebman & Aucott, 2020).
| Number in Favor | Number Opposed | Number Abstained | Number Absent |
|---|---|---|---|
| 8 | 0 | 0 | 0 |
Minority Responses
There were no minority responses.
Finding 1.4. In alignment with NIH Strategic Plan for Tickborne Disease Research, fund, support, and encourage CBPR for PLD/CLD and complex presentations of late Lyme disease, including the development and growth of community research capacity to accelerate the fundamental knowledge base using “big data” registries, data-sharing platforms, specimen and tissue sample repositories, and genomic and precision medicine approaches that reflect the underlying heterogeneous nature of PLD/CLD. This effort may include leveraging existing NIH resources and expanding collaborations across NIH Institutes to promote a multidisciplinary approach to TBD research with, for example, the NIH Community-Based Participatory Research Program (https://www.nimhd.nih.gov/programs/extramural/community-based-participatory.html).
| Number in Favor | Number Opposed | Number Abstained | Number Absent |
|---|---|---|---|
| 8 | 0 | 0 | 0 |
Minority Responses
There were no minority responses.
Finding 1.5. In alignment with NIH Strategic Plan for Tickborne Disease Research, utilize TBDs as a use case in the catalyzation of innovative change across Federal agencies to accelerate the pace of break- throughs to transform medicine and health such as creating capabilities to include agnostic platforms
that could be applied across many diseases (e.g., ARPA-H, or projects researching long tail diseases such as COVID, Myalgic Encephalomyelitis/Chronic Fatigue Syndrome, and Lyme disease).
| Number in Favor | Number Opposed | Number Abstained | Number Absent |
|---|---|---|---|
| 8 | 0 | 0 | 0 |
Minority Responses
There were no minority responses.
Finding 1.6. HHS, through for example the Agency for Healthcare Research and Quality and the Health Resources and Services Administration training grants, should explore opportunities to leverage telementoring to equip more primary care practitioners to deliver high-quality care to the PLD/CLD community (Centers for Disease Control and Prevention, 2021b).
Telementoring platforms such as Project ECHO have demonstrated significant impact on improving access to quality care for underserved and/or stigmatized populations through the use of video- conferencing technology to leverage a single specialist to contribute to increasing primary care capacity to deliver quality care. Utilizing this hub and spoke design, specialists can develop an ongoing, collaborative, telementoring relationship with clinicians across the country, which includes reach-back capability to discuss complex cases. This unique capacity to develop primary care clinicians where patients live (e.g., geographic areas with limited specialists) is a primary contributor to the powerful effects of telementoring. It has expanded over time to a number of Superhubs and Initiatives (e.g., Diabetes and Endocrinology; Opioid and Pain Related ECHOs; and Access to Specialty Care; HIV, TB, Health Security), but to date TBDs have not been included in Project ECHO’s ongoing collaboration with HHS (for example, Assistant Secretary for Preparedness and Response, Agency for Healthcare Research and Quality, and Indian Health Service (Agency for Healthcare Research and Quality, 2020; Project Echo, n.d.-a, n.d.-b ).
| Number in Favor | Number Opposed | Number Abstained | Number Absent |
|---|---|---|---|
| 8 | 0 | 0 | 0 |
Minority Responses
There were no minority responses.
Finding 1.7. In alignment with NIH Strategic Investments in Diversity, Equity, and Inclusion, NIH will identify and fund research investigating the influence of sex, race, ethnicity, pregnancy status, and socioeconomic status on the clinical manifestations of Lyme disease and other TBDs, the accuracy of diagnostic testing, and therapeutic responses.
| Number in Favor | Number Opposed | Number Abstained | Number Absent |
|---|---|---|---|
| 8 | 0 | 0 | 0 |
Minority Responses
There were no minority responses.
Discussion and Big Picture Summary
Patients with TBDs, and PLD/CLD in particular, experience significant health inequities due to longstanding policies and processes that often exclude meaningful patient participation in decisions that bear directly on their lives. Limited access to qualified clinicians remains a significant problem for many TBD patients, but especially those with PLD/CLD. Traditional payer models are ill-suited to equitably pay clinicians for the time spent caring for patients with PLD/CLD. Most clinicians do not accept health insurance, and some have been forced out of provider networks, putting care out of reach for many patients.
The limited availability of knowledgeable clinicians treating patients with PLD/CLD has produced a supply and demand imbalance. As overall case numbers continue to escalate, cases of PLD/CLD will also rise. Therefore, the current shortage of clinicians who treat PLD/CLD needs to be addressed urgently to improve health equity for this marginalized population.
A survey of treating clinicians identified several factors that challenge practicing in this area of medicine, including scientific uncertainty, professional stigma, and professional risks from regulatory actions. As discussed in this report, educational policies, processes, and practices are one of the root causes of the access to care and professional barriers with which patients and clinicians must contend. The existence of significant scientific uncertainty and the lack of acknowledgement of it on the CDC and NIH websites and in the CDC educational modules contribute substantially to these barriers.
Research, both basic science and clinical, is the primary long-term solution to overcoming scientific uncertainty and reducing health disparities for patients. Patients, clinicians, and researchers would benefit from the discovery of accurate diagnostic biomarkers for all TBDs, including the potential etiologies of PLD/CLD, that are clinically valid across the entire spectrum of patients. This advance would reduce diagnostic uncertainty, thereby decreasing diagnostic delays, and make it easier to design and conduct clinical trials, which could lead to more successful therapeutic regimens. Until such a diagnostic is developed, however, clinicians who diagnose Lyme disease will continue to face considerable uncertainty.
The most effective short-term solution is to teach to the scientific uncertainty—to identify where it lies and how it impacts clinical diagnostic and therapeutic decisions. The content of the CDC/NIH websites and the CDC TRAIN CME modules pertaining to Lyme disease should represent the primary literature as it is—both strengths and weaknesses—and identify scientific gaps. It should also inform clinicians of the divergent scientific perspectives regarding PLD/CLD, thereby opening the door to the use of clinical judgment and shared decision-making. Adopting these changes would reduce the pressure on clinicians who currently treat PLD/CLD, increase the knowledge base of front-line clinicians seeking
guidance to inform their practice, and provide a more hospitable environment to attract new clinicians to care for this growing patient population.
Limitations
In preparing this report, the subcommittee faced several important obstacles. Chief among these was the compressed time frame in which to accomplish the task. Although the subcommittee sought to more fully understand the structural determinants of health pertaining to patients with a TBD, the subcommittee was unable to speak with people who represent important perspectives such as insurers, independent medical reviewers, and researchers who could not secure NIH grants.
Appendix 1: Methods
Characteristics of the Subcommittee
The purpose of this section is to provide the methods used by the Access to Care and Education Subcommittee to prepare its report to the Tick-Borne Disease Working Group.
Membership
The Subcommittee consisted of nine members including two Co-Chairs, one from the Federal and one from the public sector. The remaining seven members included a Federal representative and six public representatives. The Subcommittee is representative of all regions of the country: Northeast, Mid- Atlantic (East Coast), Midwest, Southwest, and West Coast. (See Table 12 for the complete list of members.)
The Subcommittee expertise is derived from the civilian and Federal sectors, including the military, and runs the gamut from people whose experience comes from their profession to those whose personal experiences drive their expertise. There are several advocates who run nonprofit organizations and may also be patients or family members of patients. Healthcare providers are also well represented on the Subcommittee, and there is public health representation as well.
Several Subcommittee members are educators of physicians/clinicians, students in university settings, and the general public. One member develops TBD CME courses for clinicians. Several educate government officials on the needs of patients and treating physicians to help shape public TBD healthcare policy. A few have experience reviewing the available scientific literature as grant reviewers for nonprofit organizations and/or government programs; one has experience reviewing and also applying for research grants; and several others have served as journal peer reviewers. Several have conducted research as well as published in peer-reviewed journals. One has a global perspective on TBD in the military, and several others have experience working within the area of government public policy and government healthcare policy.
Subcommittee Meetings and Report Development and Adoption
The Subcommittee scheduled a total of 14 meetings from late September 2021 until early February 2022 (Table 2). The Co-Chairs reviewed the recommendations from previous reports as well as progress to date in achieving them, to ensure that relevant, outstanding items were addressed in scope of this report. The Co-Chairs identified areas that required expert presenters to inform the report and proposed a list of those presenters as well as a timeline for developing the report (see Table 12). The Co- Chairs then envisioned and vetted the concept of framing the report around Health Equity, which was embraced by the members. After hearing from the expert presenters, the Co-Chairs, as well as members, divided the content areas and developed sections of the report. One Co-Chair took the lead in merging all the independent sections into a final report, ensuring its presentation with one voice and internal consistency. A list of findings was developed and examined one-by-one where members refined the verbiage to ensure clarity and consistency, as well as to ensure actionability and direction to appropriate agencies and organizations where possible. The Subcommittee unanimously voted to adopt all findings. These findings, as well as the unanimously adopted entire report, will be presented to the entire Tick-Borne Disease Working Group at its next meeting.
Literature Selection
The literature used to support the data presented in this report was selected from peer-reviewed journals, expert presentations to the Subcommittee, and from Subcommittee members themselves who have relevant backgrounds and experience related to the content they have written. Additionally, the entire Background, Results, and Findings sections of the report were thoroughly reviewed and discussed by the Subcommittee members, who often added other citations to strengthen the content. The Subcommittee Co-Chairs, and several Subcommittee members, scrutinized the final report to ensure that as many relevant and up-to-date citations as possible were included.
Public Comment and Inventory
The Public Comments Subcommittee summarized and categorized all public comments received by the Tick-Borne Disease Working Group and sent them to the Subcommittee Co-Chairs to review and distribute to the Subcommittee. A Co-Chair analyzed the comments pertaining to this Subcommittee and categorized them as either already addressed, not needing to be addressed, or not pertaining to the subcommittee’s work.
Brief for the Working Group
The Subcommittee Co-Chairs prepared a PowerPoint presentation that frames Health Equity as the priority for the Subcommittees report as well as the seven findings that were voted on and unanimously approved by the Subcommittee. The presentation was shared with the Workgroup with a request for any suggested revisions, enhancements, or deletions of material. Any suggested revisions, enhancements, and deletions were considered by the Co-Chairs and applied, as appropriate, to the presentation. The presentation was used to brief the entire Tick-Borne Disease Working Group at the meeting on February 28 and March 1, 2022.
| Members | Stakeholder Group | Expertise | |
|---|---|---|---|
| Co-Chairs | |||
| Elizabeth (Betty) Maloney, MD | Public | Provider Educator Patient Advocate | Physician educator providing CME courses on Lyme disease and other TBDs Authored GRADE-based clinical practice guidelines on Lyme disease |
| Rebecca Bunnell, MPAS, PA-C, Captain, United States Public Health Service | Federal | Federal Employee, Provider, Public Health | Senior Advisor, Division of Model Learning Systems, Learning and Diffusion Group, The Center for Medicare and Medicaid Innovation, Centers for Medicare and Medicaid Services |
| Members | |||
| Holly Ahern, MS, MT(ASCP) | Public | Educator, Patient Advocate, Researcher | Associate Professor of Microbiology; Educator, patient advocate, researcher, and scientific advisor for Focus on Lyme Foundation |
| Megan DuLaney, MS | Public | Patient Advocate | Senior Advisor in support of U.S. Department State; Public/Patient, Patient experience; military health, global health, policy development and analysis |
| Lorraine Johnson, JD, MBA | Public | Patient Advocate Researcher | Principal Investigator of the patient-led research project entitled MyLymeData; subject matter expert in patient-centered healthcare policy, big data, patient registries and innovative research. Chief Executive Officer, MyLyme.Org |
| David Roberts, MD | Public | Provider Educator Researcher | Associate Professor of Medicine; Dean for External Education, Harvard Medical School. Educator for clinicians, trainees, and the public |
| Ginger Savely, DNP* | Public | Provider Patient Advocate Researcher | Provider specializing in the treatment of Lyme disease; Member of ILADS; Speaker and author with a focus on Lyme disease |
| Leith J States, MD, MPH, MBA, FACPM | Federal | Federal Employee Provider Public Health | Chief Medical Officer, Office of the Assistant Secretary for Health, Department of Health and Human Services |
| Beatrice Szantyr, MD | Public | Provider Educator Patient Advocate | Physician educator on Lyme and other TBDs. Serves on the Maine CDC Vector Borne Work Group |
* Departed the Subcommittee on January 24, 2022
| Meeting No. | Date | Present | Topics Addressed |
|---|---|---|---|
| 1 | September 20, 2021 | Rebecca Bunnell Elizabeth Maloney Holiday Goodreau Linden Hu Holly Ahern Megan Dulaney Lorraine Johnson David Roberts Ginger Savely Leith States Beatrice Szantyr |
Introductions, outlined/discussed priorities, assignment of topics, discussion of potential presentations |
| 2 | October 5, 2021 | Rebecca Bunnell Elizabeth Maloney Holly Ahern Lorraine Johnson David Roberts Leith States Beatrice Szantyr James Berger, DFO |
Discussion on potential guest speakers, Lyme disease education, and general strategy for developing the SC report. Lorraine Johnson also updated the group on the progress of the Clinician Survey. |
| 3 | October 19, 2021 | Rebecca Bunnell Elizabeth Maloney Holiday Goodreau Linden Hu Holly Ahern Megan Dulaney Lorraine Johnson Ginger Savely Beatrice Szantyr James Berger, DFO |
The DFO provided updates, contractors were introduced, speaker presentations/topics including, advocacy issues were discussed, and action items were identified and assigned. Presentation by Holly Ahern, MS, MT(ASCP) on Lyme disease and women’s health. |
| 4 | November 2, 2021 | Rebecca Bunnell Elizabeth Maloney Holiday Goodreau Holly Ahern Megan Dulaney Lorraine Johnson David Roberts Leith States Beatrice Szantyr James Berger, DFO |
Presentations by Sam Perdue, PhD on the NIAID Grants Process; and by Jeremy Richards, MD, MA, FACP, ATSF on Teaching Uncertainty in Clinical Reasoning |
| 5 | November 16, 2021 | Rebecca Bunnell Elizabeth Maloney Linden Hu Holly Ahern Megan Dulaney Lorraine Johnson David Roberts Ginger Savely Leith States Beatrice Szantyr James Berger, DFO |
Presentations by Sue Partridge, MPH and Paul Mead, MD, MPH on Tick-borne Disease Communication; Presentation by Ginger Savely, DNP on Barriers to Treatment of Lyme Disease |
| 6 | November 30, 2021 | Rebecca Bunnell Elizabeth Maloney Linden Hu Holly Ahern Megan Dulaney Lorraine Johnson David Roberts Ginger Savely Leith States Beatrice Szantyr James Berger, DFO |
Presentations by Angel Davey, PhD on Congressionally Directed Medical Research Program (DMRP) Tick-Borne Disease Research Program (TBDRP); and by Gordon Guyatt, MD on Clinical Practice Guidelines: Decisions under Uncertainty. |
| 7 | December 14, 2021 | Rebecca Bunnell Elizabeth Maloney Linden Hu Holly Ahern Megan Dulaney David Roberts Leith States Beatrice Szantyr James Berger, DFO |
Presentations by Richard Schwartzenstein, MD on Uncertainty in Medicine; by Miranda Lynch-Smith on Health Equity; and by Mary Nix on AHRQ National Guideline Clearinghouse |
| 8 | December 28, 2021 | Rebecca Bunnell Elizabeth Maloney Linden Hu Holly Ahern Megan Dulaney Lorraine Johnson Ginger Savely Leith States Beatrice Szantyr James Berger, DFO |
Presentation by Ruth Martinez, MA, CMBE on Minnesota Board of Medical Practice; discussion on organization of Subcommittee report |
| 9 | January 4, 2022 | Rebecca Bunnell Elizabeth Maloney Linden Hu Holly Ahern Megan Dulaney Lorraine Johnson Beatrice Szantyr James Berger, DFO |
Discussion of Subcommittee Report including outstanding issues, the background and methods sections, and incorporation of materials including recommendations and vignettes. The Subcommittee also heard updates on the clinical survey, and discussed plans for any additional speakers. |
| 10 | January 11, 2022 | Rebecca Bunnell Elizabeth Maloney Linden Hu Holly Ahern Megan Dulaney Lorraine Johnson Leith States Beatrice Szantyr James Berger, DFO |
Discussion of the Subcommittee Report including: outstanding issues, how to frame the findings, how the findings could get enacted, how the report could frame access to care and education, the role of advocacy, how to frame critiques of CDC/NIH. The Subcommittee also discussed how to maintain momentum post-report. |
| 11 | January 18, 2022 | Rebecca Bunnell Elizabeth Maloney Holly Ahern Megan Dulaney Lorraine Johnson David Roberts Beatrice Szantyr James Berger, DFO |
Betty Maloney, MD, led the Subcommittee discussion on progress on various sections of the report, as well as findings/potential action items for the TBDWG to consider as recommendations. |
| 12 | January 25, 2022 | Elizabeth Maloney Holly Ahern Megan Dulaney Lorraine Johnson Leith States Beatrice Szantyr James Berger, DFO |
Betty Maloney, MD, led the Subcommittee discussion on progress on various sections of the report, as well as findings/potential action items for the TBDWG to consider as recommendations. |
| 13 | February 1, 2022 | Rebecca Bunnell Elizabeth Maloney Holly Ahern Megan Dulaney Linden Hu Lorraine Johnson Leith States Beatrice Szantyr James Berger, DFO |
Betty Maloney, MD, led the Subcommittee discussion on progress on various sections of the report and voted on findings. |
| 14 | February 8, 2022 | Rebecca Bunnell Elizabeth Maloney Holly Ahern Megan Dulaney Linden Hu Lorraine Johnson Leith States Beatrice Szantyr James Berger, DFO |
Betty Maloney, MD, led the Subcommittee discussion reviewing progress on various sections of the report and the vote on the outstanding sections of the report. |
| Meeting No. | Presenter(s) | Topics Addressed | Ok to Share? |
|---|---|---|---|
| 1 | n/a | n/a | n/a |
| 2 | n/a | n/a | n/a |
| 3 | Holly Ahern, MS, MT(ASCP) | Lyme Disease and Women’s Health Lyme Disease and Women’s Health: Sex and Gender, Differences in Lyme Disease Testing, Symptoms, and Treatment | Yes |
| 4 | Sam Purdue, PhD; Jeremy B. Richards, MD, MA, FACP, ATSF | NIAID Grants Process guidelines, rates of success, and best practices in seeking research funding through NIH/NIAID. Teaching Uncertainty in Clinical Reasoning Teaching clinical reasoning skills, including the theory behind unconscious reasoning, decision certainty analysis tests (DCAT), abstract-based vignettes, and gamified clinical reasoning exercises | Yes |
| 5 | Sue Partridge, MPH, and Paul Mead, MD, MPH; Ginger Savely, DNP | Tick-borne Disease Communication Overview of how CDC communicates information to the public on tick-borne diseases. Barriers to Lyme disease treatment Overview of clinician experience in treating Lyme patients | Yes |
| 6 | Angel Davey, PhD; Gordon Guyatt, MD | Congressionally Directed Medical Research Program on Tick Borne Disease CDRMP structure, goals, and program cycle. She also highlighted the TBDRP, which is the tick-borne disease research program within CDRMP. Clinical Practice Guidelines: Decisions under Uncertainty Clinical practice guidelines and how to make decisions in uncertain situations. |
Yes |
| 7 | Richard Schwartzstein, MD; Miranda Lynch-Smith; and Mary Nix | Uncertainty in Medicine: Helping doctors and patients deal with the unknown Diagnosis vs hypothesis, cognitive biases, conversations with patients about uncertainty. Equity Briefing for the Tick-Borne Diseases Working Group Subcommittee on Access and Education How structural and social determinants of inequity lead to individual and community level disparities. | Yes |
| 8 | Ruth Martinez, MA, CMBE | Board of Medical Practice: Authority, Mission & Process Overview of organization, processes, and dealing with complaints about providers. | Yes |
| Meeting Number or Date | Motion | Result In Favor | Result Opposed | Result Abstained | Result Absent | Minority Response |
|---|---|---|---|---|---|---|
| 13 | Finding 1.1 | 8* | 0 | 0 | 0 | 0 |
| 13 | Finding 1.2 | 8* | 0 | 0 | 0 | 0 |
| 13 | Finding 1.3 | 8* | 0 | 0 | 0 | 0 |
| 13 | Finding 1.4 | 8* | 0 | 0 | 0 | 0 |
| 13 | Finding 1.5 | 8* | 0 | 0 | 0 | 0 |
| 13 | Finding 1.6 | 8* | 0 | 0 | 0 | 0 |
| 14 | Finding 1.7 | 8* | 0 | 0 | 0 | 0 |
| 14 | Background Section of the Report | 8* | 0 | 0 | 0 | 0 |
| 14 | Results Section of the Report** | 8* | 0 | 0 | 0 | 0 |
*One member was not present for the meetings 13 and 14 but reviewed and voted in favor of Findings 1.1-1.7, as well as approved the Background and Results Sections of the Report via email.
**A subsequent vote to remove a portion of Appendix 2 was approved by 7 members, no opposed, and 1 absent.
Appendix 2: Illustrative Clinician Experiences
Although the following clinician statements are posted as being anonymous, their identities are known to one of the Subcommittee Co-chairs.
Clinician 1
The first indication of trouble arrived unexpectedly, by registered letter. Reading in disbelief, I learned that an insurance company’s credentialing committee had reviewed a quality of care case concerning my treatment of Lyme disease and, without ever speaking to me, determined that ”I provided inappropriate diagnosis and treatment of tick-borne illnesses”. The letter went on to inform me that I was terminated from the insurer’s provider network. No details regarding the care concerns were provided.
Exclusion for the network would be a death blow to my practice, which provided the full range of primary care services in a small community where this particular insurer dominated the market. While practices that solely treat tick-borne diseases are commonly cash-based, patients with other health needs typically seek out clinicians that will accept their insurance. Because the proposed “remedy” appeared to extend well beyond the contours of the question of Lyme disease diagnosis and management, I decided to ask for a reconsideration of the credentialing committee’s decision. The legal fees associated with this request were $8359.60.
Although the letter did not identify who reported the quality of care concern, I knew that it did not originate from one of the patients I was treating for Lyme disease. On the contrary, the patients were pleased with the improvements they made under my care and several patient letters were included in my response to the credentialing committee.
The identity of the credentialing committee members and their knowledge of tick-borne illnesses were also unknown to me. I, on the other hand, disclosed that I had: 1) completed 137.5 hours of American Medical Association accredited Continuing Medical Education credits on the topics of Lyme disease, tick- borne illnesses, and other associated diseases; 2) done extensive review of the applicable peer reviewed literature; and 3) remained up to date on recent developments in diagnosis and treatment based on rigorous studies.
My letter and supporting documentation exceeded 700 pages; except for the patient letters, the content was comprised of papers from the peer-reviewed medical literature. Three months later, the
committee’s letter of reconsideration arrived – I must agree to treat Lyme patients in a prescribed manner or I would be removed from the insurer’s network.
And so, I was forced to pick between the small group of marginalized, suffering Lyme disease patients that I had helped and my non-Lyme disease patients who made up the bulk of the practice. With a heavy heart, I chose the latter.
Clinician 2
In 2001, I was inspired to embark on a comprehensive study of Lyme disease and its diagnosis and treatment due to my daughter’s experience with the infection. I was mentored by a Lyme specialist in the state and my collaborating physician was supportive of my work.
As patients from across my state and region began seeking my care, I came to the attention of local infectious disease (ID) physicians who disapproved of a nurse practitioner using diagnosis and treatment methods that challenged the prevailing paradigm. Despite evidence to the contrary, some local physicians were convinced that Lyme disease did not exist in their state and they promoted the idea that a short course of doxycycline is sufficient therapy for patients who acquire the infection elsewhere.
Many patients with typical symptoms of Lyme disease who had consulted these ID physicians were told they were simply depressed and needed psychotherapy. When these same patients later saw me, they were treated with antibiotics and improved. From 2003 to 2006, at the instigation of the state Board of Medical Examiners (BME), I endured a drawn-out investigation by the state Board of Nurse Examiners (BNE). Without warning, officers of the BNE arrived at my office and demanded that I immediately turn over multiple documents; not an easy task in the midst of a busy clinic day. I felt like a criminal who was being raided by the FBI. I was forced to hire an attorney who viewed the mounting allegations as harassment. Ironically, during this 3-year ordeal I was honored as my state’s Nurse Practitioner of the Year.
The BNE’s investigation produced no significant findings and I paid only a $200 fine. However, attorney fees and the intense stress of the experience left me scarred. Toward the end of the investigation, my collaborating MD was warned by the executive director of the BME that he was putting himself at risk by working with me. Although he trusted my clinical knowledge and skills, he couldn’t risk losing his license and livelihood, so he reluctantly ended our professional relationship.
In my state, nurse practitioners are required to practice in collaboration with MDs. Although many local physicians referred patients to me, none would agree to collaborate with me out of fear of the state BME. Without a collaborating MD, I couldn’t practice. Thus, the state BME essentially ran me out of the state, leaving most of my patients stranded without ongoing access to care for their Lyme disease.
Clinician 3
I was initially the subject of a Utilization Review inquiry by a regional health carrier in 2000. The carrier identified a dozen cases of Lyme disease patients who had received intramuscular penicillin. None of the patients were identified because of an adverse event, poor outcome, or malpractice. I wrote three referenced responses to their inquiry and the investigation was discontinued.
The following year the state medical board launched an investigation of the same cases initially identified by the regional carrier. I was interviewed in 2002 by an investigator at the Health Department. The medical board did not seek any action.
After receiving two additional cases, the medical board reopened the investigation in 2011. Every case had been diagnosed and treated for Lyme disease. None of the patients were identified because of an adverse event, poor outcome, or malpractice. The investigator focused on the physical examination and differential diagnosis.
The prosecutor verbally offered my attorney a six-month suspension of my license followed by probation for a period of 2.5 years under specified terms and conditions for professional misconduct. Because the attorney was unable to negotiate better terms with the prosecutor, I asked the courts for a dismissal of the charges. Despite accruing more than $500,000 in legal fees, I lost the appeal.
Ultimately, I signed a consent order for misconduct related to problems with taking an adequate history, performing an appropriate physical examination, and failing to construct differential diagnoses and conduct a thorough diagnostic evaluation. The agreement allowed me to practice medicine during my three-year probationary period but required that I be closely supervised by a physician monitor.
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