Chronic Lyme Disease: A Survey of Connecticut Primary Care Physicians

Michael Johnson, MD and Henry M. Feder, Jr. MD

Objective To determine how frequently Connecticut primary care physicians are diagnosing and treating patientswith chronic Lyme disease.Study design A survey was mailed to a random 33% sample of primary care physicians in Connecticut, whichasked how many cases of Lyme disease and chronic Lyme disease they diagnosed and treated in the last 3 years.Results The survey had a response rate of 39.1%. Physician respondents (n = 285) fit in one of 3 groups. Group 1included 6 of 285 (2.1%) physicians who diagnose and treat patients with chronic Lyme disease. Group 2 included137 of 285 (48.1%) physicians who are undecided on the existence of chronic Lyme disease, but did not diagnose ortreat any patients with chronic Lyme disease. Group 3 included 142 of 285 (49.8%) physicians who do not believe inthe existence of chronic Lyme disease.Conclusion A small percentage (2.1%) of primary care physicians diagnose and treat patients for presumedchronic Lyme disease. (J Pediatr 2010;157:1025-9).

The existence of ‘‘chronic Lyme disease’’ has been sharply debated in the last decade.1-3 Chronic Lyme disease has beendefined by its supporters as persistent Borrelia burgdorferi infection requiring months to years of intravenous antibiotictherapy, oral antibiotic therapy, or both.4 Prolonged antibiotic therapy for presumed chronic Lyme disease is not rec-

ommended by the American College of Rheumatology,5 the American Academy of Pediatrics,6 the Infectious Diseases Societyof America (IDSA),7 or the American College of Neurology.8 The IDSA Lyme disease treatment guidelines specifically warnagainst using intravenous antibiotic therapy for presumed chronic Lyme disease.7 Opposing the recommendations from thesemedical societies is a well-organized network of physicians, patients, and patient advocates who, via testimonials, case histories,and other anecdotal forms of evidence, promote the concept of chronic Lyme disease in the medical literature,9,10 in the layliterature,11-13 and on the Internet.14

The chronic Lyme disease advocates have started their own society (International Lyme and Associated Diseases Society[ILADS]) and published their own Lyme disease diagnostic and treatment guidelines.4 These advocacy groups, which referto themselves as ‘‘Lyme literate,’’ include physicians, patients, and laboratory personnel.15 Protests have occurred when insur-ance companies will not pay for prolonged (90-day) courses of intravenous antibiotics prescribed for patients with presumedchronic Lyme disease.1 As a result, chronic Lyme disease advocates have initiated legislation requiring insurance companies topay for prolonged intravenous antibiotic therapy (Connecticut House Bill 5625, 2009). An argument put forth by advocates forchronic Lyme disease is that a large number of community-based primary care physicians do not support or follow the aca-demic guidelines of the American Academy of Pediatrics, IDSA, and American College of Neurology. Instead, thesecommunity-based physicians commonly diagnose patients with chronic Lyme disease and treat these patients with prolongedcourses of intravenous antibiotics, oral antibiotics, or both.1 The purpose of our study was to survey a random sample of Con-necticut primary care physicians to determine whether they diagnose and treat patients with what they believe is chronic Lymedisease.

Lyme Disease

Lyme disease is a multisystem infection caused by the spirochete Borrelia burgdorferi. The clinical manifestations were initiallydivided in stages, but now are divided in early localized, early disseminated, and late Lyme disease.7,16,17 Early localized Lymedisease is characterized by the rash erythema migrans (EM). Early disseminated Lyme disease is characterized by multiple er-ythema migrans, cardiac findings (usually carditis with second- or third-degree heart block), neurologic findings (usually cra-nial neuritis, lymphocytic meningitis, or motor/sensory radiculoneuritis). Late Lyme disease is characterized by rheumatologicfindings (usually arthritis involving the knee) or objective neurological findings (usually neuropathy, encephalitis, or enceph-alopathy). Lyme disease usually can be treated with 10 to 28 days of a single oral antibiotic. Patients with meningitis, enceph-alitis, encephalopathy, or carditis may require 14 to 28 days of intravenous antibiotic therapy.6,7 Patients with Lyme disease

From the University of Connecticut Health Center,Farmington, CT (M.J.); and Departments of Pediatricsand Family Medicine, University of Connecticut HealthCenter, Farmington, CT (H.F.)

The authors declare no conflicts of interest.

0022-3476/$ - see front matter. Copyright � 2010 Mosby Inc.

All rights reserved. 10.1016/j.jpeds.2010.06.031

EM Erythema migrans

IDSA Infectious Diseases Society of America

ILADS International Lyme and Associated Diseases Society

SD Standard deviation

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Physicians who diagnose and treat chronic Lyme diseasePhysicians who are undecided with respect to chronic Lyme diseasePhysicians who do not believe chronic Lyme disease exists

Figure 1. The practices of Connecticut primary care physi-cians for chronic Lyme disease.

THE JOURNAL OF PEDIATRICS � www.jpeds.com Vol. 157, No. 6

need to have visited an area where Lyme disease is endemic.Patients with objective cardiac, neurologic, or rheumatologicmanifestations of Lyme disease should have positive B burg-dorferi serology results. Seropositivity is defined by positiveresults on enzyme-linked immunosorbent assay and a posi-tive immunoblot test result. Serology may remain positivefor years after successful treatment of Lyme disease. Manyother unvalidated tests are available (including urine tests)to presumably document acute and chronic B burgdorferi in-fection.7,16-18

ConnecticutIn 1977, Steere et al19 reported a cluster of patients (39 chil-dren and 12 adults) with a mysterious arthritis. The patientscame from 3 bordering Connecticut communities, OldLyme, Lyme, and Haddam. The illness was named Lyme ar-thritis. In the next decade, the mysteries of Lyme arthritiswere solved. Lyme disease or Lyme borreliosis became recog-nized as a deer tick-borne multisystem infection caused bythe spirochete B burgdorferi. According to the Centers forDisease Control and Prevention, Connecticut has the highestincidence of Lyme disease in the United States.20,21

Chronic Lyme diseaseChronic Lyme disease has been defined by ILADS as a chronicdebilitating illness with subjective symptoms such as fatigue,arthralgia, myalgia, poor concentration, headaches, and irri-tability.4 ILADS states that chronic Lyme disease is caused bypersistent infection with B burgdorferi unresponsive to stan-dard courses of antibiotic therapy. Patients with chronicLyme disease may or may not have a history of objective find-ings consistent with Lyme disease, may or may not have pos-itive B burgdorferi serology results, and may or may not havevisited an area where Lyme disease is endemic. According toILADS, patients with chronic Lyme disease need months toyears of treatment with oral antibiotics, intravenous antibi-otics, or both (frequently with multiple antibiotics simulta-neously). Even after years of treatment, B burgdorferiinfection may persist. Diagnosing and treating chronicLyme disease is based on the judgment and experience ofthe treating physician.4

Methods

A list of physicians who were licensed in Connecticut in 2006was obtained from the Connecticut Department of PublicHealth (Health Care Licensing Database). We searched thislist for primary care physicians who practiced family medi-cine, internal medicine, or pediatrics. Physicians from thisgroup who listed a subspecialty or who practiced outsideConnecticut were eliminated. Our search yielded 3091 physi-cians. A 33% sample of the 3091 physicians was chosen ran-domly with a number generator in Microsoft Excel 2003(Microsoft Corp., Redmond, Washington). Only one mailingwas done in this study. Surveys that were returned did notspecifically identify the physician responder. Thus, non-responders could not be sent a second mailing.

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A 1.5-page survey (Appendix; available at www.jpeds.com) was mailed to each physician. As background,physicians were told that chronic Lyme disease wasa controversial topic and that it is was unknown howfrequently primary care physicians diagnosed it and treatedpatients with chronic Lyme disease. Chronic Lyme diseasewas defined as persistent B burgdorferi infection requiringprolonged antibiotic therapy; furthermore, patients withchronic Lyme disease may never have had objective signsconsistent with Lyme disease and may be seronegative for Bburgdorferi antibodies.

Physicians were asked whether they were actively practic-ing family medicine, internal medicine, and/or pediatricsand whether they were practicing a subspecialty. Physicianswere then asked to estimate the number of their primarycare patients in whom they had diagnosed and treatedLyme disease, chronic Lyme disease, or both in the last 3years. They were also asked about the length of antibiotictherapy that they prescribed for their patients with Lyme dis-ease and chronic Lyme disease. For chronic Lyme disease,physicians were asked whether they were experienced in diag-nosing it and treating patients with chronic Lyme disease,whether they were undecided as to the existence of chronicLyme disease, or whether they felt that chronic Lyme diseasedid not exist. At the end of the questionnaire, there wasa space for comments.

An average and SD was calculated for the entire data set forthe number of Lyme disease and chronic Lyme disease casesdiagnosed by each physician. A z test was done, and outliers 3SDs from the mean (z > 3.0) were removed from the data setwhen comparing the number of Lyme cases diagnosed perphysician per year in groups. The University of Connecticutinstitutional review board approved the study. The re-sponders and non-responders could not be identified, andthus non-responders could not be sent a second mailing.

Johnson and Feder Jr.

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Figure 2. Cases of Lyme disease and chronic Lyme diseasediagnosed in the past 3 years by 6 physicians who believe thatchronic Lyme disease exists.

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Figure 3. Cases of Lyme disease diagnosed in the past 3years by A, 137 physicians who are undecided with respect tochronic Lyme disease and B, 142 physicians who do not be-lieve chronic Lyme disease exists.

December 2010 ORIGINAL ARTICLES

Results

A total of 1034 surveys were mailed to physicians’ offices, and191 were returned because of outdated or incorrect ad-dresses; thus 843 surveys were successfully mailed. Threehundred thirty responses were received, for a 39.1% (330/843) response rate. Of the 330 surveys, 45 respondents wereomitted from this analysis: 10 were from physicians no longerin practice; 2 were not from physicians; 20 were from physi-cians with a subspecialty; 5 were from physicians who had notdiagnosed Lyme disease or chronic Lyme disease in any pa-tients in the last 3 years; and 8 had undecipherable answers.

Of the 285 responses included in the study (Figure 1), 6were from physicians (2.1%) who diagnosed chronic Lymedisease and treated patients with it (group 1). One hundredthirty-seven physicians (48.1%) were undecided as to theexistence of chronic Lyme disease (group 2). One hundredforty-two physicians (49.8%) felt chronic Lyme disease didnot exist (group 3). Of the physicians in group 1, 50% werefamily physicians and 50% were internists; in group 2, 21%were family physicians, 46% were internists, and 28% werepediatricians; in group 3, 17% were family physicians, 33%were internists, and 48% were pediatricians. A fewphysicians in groups 2 and 3 were emergency or medicine/pediatrics physicians.

In the last 3 years, the 285 physicians in our survey esti-mated that they had diagnosed 11 970 cases of Lyme diseaseor 14 cases of Lyme disease per physician per year (outliersincluded). In addition, 6 physicians estimated that they haddiagnosed 84 cases of chronic Lyme disease or 4.7 cases ofchronic Lyme disease per physician per year. Histograms de-picting the number of cases diagnosed per physician weremade for each group and are shown in Figures 2 and 3.The histograms demonstrate outliers in each group. Fiveoutliers (>3 SDs above the mean) were included thatdiagnosed $100 cases of Lyme disease each year: one fromgroup 1, two from group 2, and two from group 3.

The 6 physicians in group 1, who diagnosed chronic Lymedisease and treated patients for it, diagnosed an average of 3.1cases of chronic Lyme disease per physician per year (whenthe outlier who diagnosed 40 cases per year was excluded).In addition, they diagnosed an average of 11.5 cases ofLyme disease (not chronic) per physician per year (whenthe outlier who diagnosed $100 cases per year was excluded).Each physician in group 1 diagnosed more cases of Lyme dis-ease than chronic Lyme disease. The average was one case ofchronic Lyme disease for every 9 cases of Lyme disease (range,2.4-18.0 cases of Lyme disease for each case of chronic Lymedisease with outlier included).

The 137 physicians in group 2, who were undecided or didnot have an opinion about the existence of chronic Lyme dis-ease, diagnosed an average of 11.4 cases of Lyme disease perphysician per year (when the two outliers who diagnosed$100 cases per year each were excluded).

The 142 physicians in group 3, who did not believe thatchronic Lyme disease exists, diagnosed an average of 12.4

Chronic Lyme Disease: A Survey of Connecticut Primary Care Ph

cases of Lyme disease per physician per year (when the twooutliers who diagnosed $100 cases per year were excluded).

The physicians in groups 1, 2, and 3 treated patients withLyme disease for 2 to 4 weeks. The physicians in group 1used an average of 20 weeks (range, 8-52 weeks) of antibiotictherapy to treat patients in whom they diagnosed chronicLyme disease.

Some of the physicians (159/279) in groups 2 and 3reported that other physicians had diagnosed chronic Lymedisease in many of their primary care patients. Most

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physicians reported that their patients in whom chronic Lymedisease was diagnosed were not helped by the oral and intra-venous antibiotics, and sometimes underlying diagnoses,such as depression and in one case leukemia, were missed.

Discussion

Our study is a large survey asking primary care physiciansabout chronic Lyme disease practices. The acceptance or re-jection of the concept of chronic Lyme disease has polarizedConnecticut physicians. Approximately half (48.1%) thephysicians (group 2) had no opinion about chronic Lymedisease. We found that only 6 of the 285 physicians (2.1%)diagnose chronic Lyme disease and treat patients with it.Five of the 6 physicians treated patients with chronic Lymedisease for <6 months; one physician treated patients withchronic Lyme disease for 12 months. No physician com-mented that he/she used intravenous antibiotics to treat pa-tients with chronic Lyme disease. Thus, the physicians weidentified who diagnose chronic Lyme disease and treat pa-tients with it differ from ‘‘Lyme literate’’ physicians becausethe ‘‘Lyme literate’’ physicians treat hundreds of patientswith chronic Lyme disease each year, with months to yearsof antibiotics given orally or intravenously. ‘‘Lyme literate’’physicians did impact our study, because 159 of the 279 phy-sicians in our groups 2 and 3 reported that many of their pri-mary care patients were diagnosed as having chronic Lymedisease by other physicians, and some of these patientswere treated with antibiotics intravenously.

The origin of chronic Lyme disease can be traced to the1989 publication by Burrascano,22 who described his per-sonal experiences treating 284 patients with Lyme disease.Of the 284 patients, 269 (95%) became chronically infectedwith B burgdorferi because they were initially treated witha <60-day course of antibiotics.22 The initial antibiotic ther-apies included doxycycline, penicillin V, amoxicillin plusprobenecid, or ceftriaxone intravenously. A few years later,Burrascano23 reported that he was following thousands of pa-tients with chronic Lyme disease who had B burgdorferi infec-tion resistant to standard therapy. In these early reports,Burrascano did not give a case definition of chronic Lymedisease and did not specifically define the treatment ofchronic Lyme disease.

In 1998, Phillips et al24 reported recovering B burgdorferifrom the blood of 39 of 41 patients (95%) with chronicLyme disease who were receiving prolonged antibiotic ther-apy. Most of these patients had seronegative results for Bburgdorferi antibodies and lacked objective findings consis-tent with Lyme disease. This study by Phillips could not bereproduced, and the culture medium used by these investiga-tors subsequently was shown to be bactericidal for B burgdor-feri.25

In the late 1990s, Burrascano, Phillips, and other physi-cians organized the ILADS society to promote national andinternational awareness of chronic Lyme disease. ILADSstates that chronic Lyme disease is being ignored by main-

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stream medicine. Physicians and patients who supportILADS have formed a network throughout the United States.This network includes physicians who practice in stateswhere Lyme disease is not endemic (for example, Floridaand Colorado). ILADS does not have a clear case definitionof chronic Lyme disease or specific treatment recommenda-tions, except that oral/intravenous antibiotic therapy may beneeded for months to years.4

In 1999, two Connecticut physicians testified at a publichearing that they were diagnosing and treating hundreds ofpatients each year with chronic Lyme disease.1 They maintainthat treatment of chronic Lyme disease requires months toyears of oral and intravenous antibiotic therapy. Later, an-other Connecticut physician testified that he diagnosesLyme disease in 999 of every 1000 patients (usually self-referred) that he sees (Connecticut public record, Sep 12,2008, testimony of Charles Ray Jones, MD). This physiciansees mainly children and treats many of them with antibioticsintravenously for chronic Lyme disease.

We know of only a small number of ‘‘Lyme literate’’ phy-sicians from Connecticut and the Northeastern states who di-agnose chronic Lyme disease and treat patients withantibiotics intravenously. However, this small number ofphysicians is influential. The ‘‘Lyme literate’’ network hasbeen pivotal in advocating legislation in multiple states re-quiring insurance companies to cover the costs of intrave-nous therapy for presumed chronic Lyme disease. Theattorney general of the state of Connecticut, with inputfrom ‘‘Lyme literate’’ network, issued a subpoena to theIDSA claiming unfair trade practices because the IDSA didnot acknowledge chronic Lyme disease in their Lyme diseasetreatment guidelines.7,26 Finally, on Apr 30, 2009, the Con-necticut House of Representatives passed legislation (HB6200) that reassured physicians who diagnose chronicLyme disease and treat it with long-term antibiotics thatthey will not be subject to any disciplinary actions.

Our study found that approximately 97% of primary carephysicians do not diagnose or treat chronic Lyme disease.We understand that although our sample was random,a sampling bias could occur. Physicians who diagnose andtreat chronic Lyme disease may not have responded to thissurvey because we are not part of their network. Or possibly,advocates for chronic Lyme disease may have been morelikely to respond so they could be counted. Also, the samecould be said of physicians who believe chronic Lyme diseasedoes not exist. We did not have approval from the institu-tional review board to contact the non-responders, andthus we were unable to define why physicians were non-re-sponders.

Our study, asking physicians to recollect an approximatenumber of patients diagnosed with Lyme disease in thepast 3 years, may be subject to recall bias. In each group stud-ied, there was significant variation in case numbers. The largeSD calculated in each group for Lyme disease cases can be at-tributed to the geographic variation in the frequency of thedisease throughout Connecticut. n

Johnson and Feder Jr.

December 2010 ORIGINAL ARTICLES

We thank Justin Radolf, MD, for reviewing our manuscript.

Submitted for publication Mar 22, 2010; last revision received May 20, 2010;

accepted Jun 17, 2010.

Reprint requests: Henry M. Feder, Jr, MD,Departments of Family Medicine

and Pediatrics, University of Connecticut Health Center, Farmington,

Connecticut 06030. E-mail: feder@nso2.uchc.edu.

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Appendix. Lyme disease survey

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Appendix. Continued

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