Background
Chronic Lyme disease (CLD) is a term that describes a constellation of persistent symptoms in patients with or without exposure to
Borrelia burgdorferi. These symptoms may include fatigue, night sweats, arthralgia, myalgia, arrhythmias, abdominal pain, nausea, diarrhea, sleep disturbance, cognitive dysfunction, irritability, depression, back pain, and headache [
1]. There is no standard definition of CLD; the Infectious Diseases Society of America distinguishes between ‘post-Lyme disease syndrome’ or ‘late Lyme disease’ in which arthralgia and other symptoms persist after documented
B. burgdorferi infection [
2], while Feder et al. [
1] categorize similar syndromes into post-Lyme disease syndrome, symptoms of unknown cause (with our without antibodies to
B. burgdorferi), and defined illnesses unrelated to
B. burgdorferi infection.
Nevertheless, patients are often are diagnosed with CLD based on nonstandard interpretations of serology or other testing that has little or no validity, or based on clinical symptoms alone [
2,
3]. CLD is diagnosed throughout the U.S. and globally, in both endemic and non-endemic areas [
1]. Individuals may make the diagnosis themselves using lists of subjective symptoms [
4,
5]. Treatment protocols can include regimens of multiple antibiotics (oral or parenteral), sometimes continuing for months or years [
2,
3]. These protocols are associated with substantial risks such as out-of-pocket expenses, patient distress, potential harm, as well as increasing risk of selecting for antibiotic resistant bacteria [
1,
6].
One survey found that 2.1% of Connecticut-based primary care physicians diagnose and treat CLD, while the majority were unsure of or did not believe in CLD [
7]. The predominant infectious diseases, pediatric, and neurology academic societies do not recognize CLD as a distinct clinical entity, while other professional and advocacy organizations argue for a broader definition of CLD focusing on clinical (non-serological) diagnosis based on reported symptomology [
3,
8]. Patient perceptions of lack of sympathy and alienation from mainstream medical institutions led to the growth of advocacy groups supporting long-term antibiotic treatment, as well as fostering ‘us vs. them’ sentiments in the advocacy groups [
8].
Qualitative methods are well suited to generate rich data about phenomena in their context and can help develop hypotheses for further research, as well as inform the development of patient-centered interventions [
9]. The purpose of this study was to gather insights about the experiences of patients identifying themselves as having CLD in the healthcare system using the dimensions of the Health Belief Model where perceived severity, susceptibility, benefits, and barriers; as well as cues to action, and self-efficacy are involved in influencing health behavior choices [
10]. We also aimed to gain insights into therapeutic and healthcare provider choices to better understand their illness representations, that is, their beliefs and expectations about an illness [
11,
12].
Discussion
Our participants reported substantial limitations in their daily lives and a decline in health status attributed to CLD. Some believed that they would never recover and may possibly die from CLD. Participants had polarized experiences in the healthcare system: negative experiences were associated with providers who were reported to be dismissive, patronizing, or condescending, while positive experiences were associated with providers reported to be optimistic, attentive, and supportive. Consultations with CAM practitioners and use of CAM therapies were common.
Our cohort included participants who were diagnosed with or self-identify as having CLD irrespective of whether they had objective evidence of previous borreliosis. Because our study aimed to better understand and describe individual and collective experiences and ideas (e.g., the perception of a disease), strict enrollment criteria utilizing serology would not be helpful. Therefore, the only criterion for enrollment was that a person identified as a CLD patient; the etiology and classification of their symptoms were not relevant.
Reported cases of Lyme disease to the Centers for Disease Control and Prevention (meeting surveillance criteria) are most common in the Northeastern United States among children aged 5–9, and adults aged 55–59, with male predominance (53%). Most reported cases (94%) occur in patients identified as White [
16]. Our study focused on adults; 75% of our cohort were female, all were White.
Most participants reported general health and wellness prior to CLD. In previous literature, health-related quality of life among CLD patients has been reported to be similar to patients with congestive heart failure and type 2 diabetes [
17]. Our participants also had a mean score of 56 on the Arizona Integrative Outcomes Scale (AIOS) for the month prior to their interview, indicating a compromised sense of well-being. Of note, Bell et al. reported similar mean AIOS scores of 51 for individuals who qualified as "psychologically distressed" on the Global Severity Index (a standard measure of psychological health) [
15].
It is possible that this psychological distress associated with severe symptomology, coupled with a lack of clear disease course and uncertainty about recovery led some to fear dying from CLD, despite CDC data indicating that Lyme disease rarely causes death [
18]. Previous studies have found an association between CLD and negative affect [
19], and mixed results regarding an association with anxiety and/or depression [
19,
20].
Our findings also provide insight into the dimensions of Leventhal’s illness representations of identity, cause, timeline, control/cure, and consequences [
11,
12]. Our patients strongly identified with the label of CLD, with many expressing relief to have a diagnosis to identify with their symptoms. Furthermore, attributing symptoms to active borreliosis, whether or not objective testing supported this diagnosis, was empowering in the sense that treatments (prolonged antibiotic courses and CAM therapies) can be directed towards the bacteria to control and possibly cure the condition. In our sample, the timeline was necessarily chronic (and often cyclical), as we selected patients identifying with CLD and not necessarily having a history of acute Lyme disease. The consequences of CLD in our cohort were remarkable, impacting social relationships, and activities of daily living, as well as perceptions of mortality and debility.
Some literature has classified CLD as ‘medically unexplained symptoms’ [
1]. Symptomatically, CLD can resemble other such conditions including chronic fatigue syndrome and fibromyalgia [
21], as well as neurologic conditions such as amyotrophic lateral sclerosis [
22] or multiple sclerosis [
23]. The lack of clear pathophysiology often results in extensive diagnostic workups and significant iatrogenic complications [
24]. Like many chronic pain conditions, depressive and anxiety disorders are prevalent and can be overlooked [
19]. At least 13% of outpatient visits can be attributed to medically unexplained symptoms [
24].
In one study, a majority of primary care physicians described their attitudes towards patients as negative and dismissing [
25] while another study found substantial discordance between patient and physician treatment goals [
26]. Previous studies of other medically unexplained conditions note that patients often identify as ‘medical orphans’ without a medical specialty home to seek treatment. Further, their symptoms are frequently labeled psychosomatic, and their stories described as ‘chaos narratives’ lacking a defined illness trajectory with uncertainty about persistence or improvement of symptoms [
27].
Our participants reported similar experiences. Congruent with our findings, Nunes et al. [
28] also found patient concerns regarding about the illness severity to predominate over concerns about symptomatic relief in a qualitative study of patients with medically unexplained symptoms. In a study of persons with fibromyalgia, LaChapelle et al. found that patients reported mixed feelings about support groups; some valued the ‘safe haven’ aspect as an encouraging environment with peers, while other patients reported discouragement from others, perceived to be "building their whole life around the disease" [
29].
As is true of many health conditions that are poorly understood and often resistant to conventional treatments, medically unexplained conditions often compels those afflicted to seek CAM therapies and providers [
30,
31] including over-the-counter products, mind-body practices, dietary modifications, acupuncture, and chiropractic [
32].
The majority (11/12) of our participants used one or more forms of CAM, and most consulted a CAM provider. To the best of our knowledge, this is the first study to report frequent CAM use among CLD patients. The high prevalence of CAM was unexpected given that recruitment occurred in general (not CAM-oriented) email lists. It is possible that patients were more open to discussing CAM in a study led by a CAM-trained investigator (AA), whereas patients often feel reluctant to discuss CAM use with conventional providers [
33].
It is possible that CLD patients are attracted to unconventional providers – those providing extensive antibiotic regimens beyond current guidelines (‘Lyme-literate medical doctors’) and/or CAM providers due to the lack of symptomatic relief, as well as a perceived lack of sympathy to the diagnosis of CLD in more conventional settings. Our participants reported greater satisfaction with providers described as being ‘open minded’ and supported the use of CAM therapies, while expressing frustration with doctors perceived as dismissive after serology ruled out Lyme disease. The extensive financial expenditures reported may be reflective of a desire for therapeutic options beyond those conventionally available [
34].
Some reported CAM therapies may be of questionable value (i.e., the Rife Machine purporting to detect ‘vibrations’ in the body to aid in diagnosis and treatment), while others may be directly harmful. For example, thujone-containing extracts of
Artemesia absinthium (wormwood) can cause seizures, rhabdomyolysis, and acute renal failure [
35], while the use of oral colloidal silver is associated with neurological deficits, diffuse silver deposition in visceral organs, and renal damage. Nevertheless, our participants did not report any adverse effects from CAM therapies.
This study has several limitations. It was restricted to one geographic area where Lyme disease is endemic (Connecticut, USA), and social awareness of CLD is prevalent. Though our sample was small, theoretical saturation was reached. Furthermore, our participants were ethnically homogenous and computer literate, and a priori, identified with CLD. Our results may therefore not apply to more diverse groups of CLD patients and those in other geographic locations.
Competing interests
The authors declare that they have no competing interests.
Authors’ contributions
AA: study design, data collection, data analysis, data interpretation, writing. LV: study design, data analysis, data interpretation, writing. RL: data analysis, data interpretation. TRW: editorial support, writing. ERC: study design, data interpretation, writing. All authors read and approved the final manuscript.