PEDIATRICS Vol. 105 No. 4 April 2000, pp. 855-857

COMMENTARY:
Differences Are Voiced by Two Lyme Camps at a Connecticut Public Hearing on Insurance Coverage of Lyme Disease

In early February 1999, I was called by the Office of the Attorney General of Connecticut and asked to testify at a February 24th public hearing on insurance coverage of Lyme disease. A bill was in the process of development that would mandate Connecticut health insurance companies to pay for long-term intravenous antibiotic therapy for children and adults with Lyme disease. At that time, I didn't realize that this public hearing was set up to contrast 2 points of view regarding the diagnosis and treatment of Lyme disease, the 2 Lyme camps (a term coined by the lay press to describe the first camp, which include academic-based physicians who use limited antibiotic therapy to treat patients with Lyme disease vs the second camp, which include community-based physicians and patient advocacy groups who believe in open-ended antibiotic therapy to treat patients with Lyme disease). I was invited as a pediatric infectious disease specialist from Connecticut Children's Medical Center along with Robert T. Schoen, MD, an adult rheumatologist from Yale University School of Medicine. We were requested to represent academic-based physicians. We had both published about the dangers of intravenous antibiotic therapy for patients with presumed Lyme disease.^1,2 The purpose of this commentary is to describe the 2 Lyme camps, to summarize the public hearing, and to emphasize that medical public policy should not be determined by advocacy groups unless there is scientific support.³

THE TWO LYME DISEASE CAMPS

The 2 Lyme camps are clearly outlined in 2 consecutive editions of Conn's Current Therapy. In the 1997 edition,⁴ Joseph J. Burrascano, Jr, MD (a community-based physician) gives his method of diagnosing and treating Lyme disease. Dr Burrascano states that Lyme disease "is an extremely complex illness that is still poorly understood." He notes that fewer than 50% of patients with Lyme disease present with the classic rasherythema migrans. The majority of patients with Lyme disease present with "nonspecific and protean symptoms" that involve multiple systems. The diagnosis of Lyme disease is made on clinical grounds (including a response to empiric antibiotics) and no currently available laboratory test is definitive. Parenteral therapy is recommended for patients with persistent symptoms unresponsive to oral therapy and for patients with severe disease. Management of Lyme disease is a "therapeutic alliance between the physician and the patient."

In the 1998 edition of Conn's Current Therapy,⁵ Allen C. Steere, MD, (an academic-based physician) gives his method of diagnosing and treating Lyme disease. Dr Steere states that >75% of patients with Lyme disease present with erythema migrans. The other patients present with objective signs involving the nervous system, heart, or joints. "Serologic testing is the most practical laboratory aid to diagnosis."⁵ To diagnose Lyme disease with nervous system, heart, or joint involvement, Borrelia burgdorferi serology should be positive. Lyme disease can usually be treated successfully with oral antibiotics, except neurological disease, which usually requires intravenous antibiotic therapy.

The dichotomy is clear (Table 1). Dr Burrascano states chronic Lyme disease is common and defines it as persistent neurologic or musculoskeletal symptoms in a patient who may or may not have objective findings and may or may not have positive B burgdorferi serology. Dr Steere states chronic Lyme disease is uncommon and defines it as persistent neurologic or musculoskeletal symptoms in a patient with objective findings and positive B burgdorferi serology. In addition, Dr Steere defines a post Lyme syndrome as persistent neurologic or musculoskeletal symptoms without objective findings in a patient with positive B burgdorferi serology. Dr Steere states that the post Lyme disease syndrome resolves spontaneously, whereas Dr Burrascano includes Steere's post Lyme disease syndrome as chronic Lyme disease.

THE PUBLIC HEARING

The first speaker at the public hearing was Matthew Cartter, MD, MPH, from the Connecticut Department of Public Health. He stated that in 1998, Connecticut had the highest reported rate of Lyme disease in the United States, 103 cases/100 000. These cases met the Centers for Disease Control and Prevention Lyme disease surveillance case definition.⁶ This set the stage for the 4 panels of speakers that would follow. The audience, for the most part, was made up of Lyme disease patient advocacy representatives.

The first panel of speakers included 2 adult patients with chronic Lyme disease (as defined by Dr Burrascano). Each told of their frustrations with the medical system and disappointment with health insurance companies who rejected claims. A third speaker told the story of her 15-year-old daughter with chronic Lyme disease. Her daughter's illness began with back pain, low-grade fever, sore throat, and fatigue. This was followed by generalized pain, shortness of breath, inability to concentrate, irritability, intolerance to noise, paresthesias, nausea, and loss of hair. Over the course of 9 months, she visited specialists at 6 teaching hospitals, had a plethora of testing (including multiple negative B burgdorferi serologies), and no diagnosis was made. She was then empirically treated for seronegative Lyme disease and her symptoms improved after an 8-week course of intravenous ceftriaxone. Her illness was then intermittent for the next 3 years, during which time she missed >1 year of school and required an additional year of intravenous antibiotic therapy. She is now well and in college. At the end of their presentation, the Connecticut policymakers asked questions about insurance rejections.

The next panel included physician representatives from 2 of Connecticut's largest health insurance companies. Both stated that prolonged intravenous therapy for Lyme disease was not recommended for children⁷ or adults.⁸ In general, it was their policy to pay for a maximum of 6 weeks of intravenous antibiotic therapy for Lyme disease. The Connecticut policy makers asked whether the insurance companies would pay for prolonged oral therapy for Lyme disease and were answered yes. The policymakers concluded that if insurance companies paid for prolonged oral antibiotics and not prolonged intravenous antibiotics, it was an issue of cost.

Next were 2 physicians from the community who believed that Lyme disease could be diagnosed without objective findings and without positive serology, and therapy should be open-ended. The first of these speakers, Steven E. Phillips, MD, referenced his research,⁹ where he cultured B burgdorferi from 43/47 patients (91%) with presumed chronic Lyme disease. These 47 patients had been treated for a mean of 3 months with intravenous antibiotics. Over 90% were seronegative and chronic Lyme disease was diagnosed based on chronic musculoskeletal or neurologic symptoms. Dr Phillips used his research to make the point that chronic Lyme disease could occur without objective findings or positive serology; however, the study by Dr Phillips⁹ has not been reproduced. The second speaker, Amiran Katz, MD, stated that chronic Lyme disease was common. Dr Katz said he was setting up a randomized, placebo-controlled study of hyperbaric oxygen for the treatment of chronic Lyme disease. Drs Phillips and Katz recognized seronegative chronic Lyme disease and had an armamentarium of laboratory tests and therapies including long-term intravenous antibiotics. They questioned published Lyme disease studies that concluded that Lyme disease is overdiagnosed and overtreated, as biased and out-of-date.^2,10 They believed that open-ended intravenous antibiotic therapy for chronic Lyme disease should be covered by insurance companies.

Dr Schoen and I were the last speakers. Dr Schoen spoke first, about the overdiagnosis and overtreatment of Lyme disease. He reviewed his study of 209 patients with presumed Lyme disease seen at the Yale University Lyme Disease Clinic.² One hundred twenty-five of the 209 (60%) patients never had Lyme disease (these patients did not have erythema migrans, objective findings, or positive serology) and many of these patients suffered treatment-related illness and disability. In contrast, the patients with confirmed Lyme disease did well. His presentation warned of the dangers of unneeded intravenous antibiotics. At the end of his presentation, he was asked by the Connecticut policymakers about prolonged antibiotic therapy for patients with chronic Lyme disease. He answered that he was participating in a large double-blinded, placebo-controlled study of 1 month of intravenous ceftriaxone versus placebo for patients with chronic Lyme disease, defined by positive serology and chronic neurologic or musculoskeletal sign. Dr Schoen emphasized that until such studies are completed, parenteral therapy for patients with chronic Lyme disease cannot be recommended.

I was the last speaker. My purpose was to tell 3 stories describing some pitfalls of diagnosis and treatment of Lyme disease. My first story involved a 15-year-old female who developed knee swelling. Her B burgdorferi serology was markedly positive and she was started on intravenous antibiotics. Her knee swelling resolved within a few days of starting antibiotics but generalized arthralgias continued over the next month. After 6 months of intravenous therapy, she was still having intermittent arthralgias and came to see me for a second opinion. Her physical examination was normal. I said she had had Lyme arthritis, which had been more than adequately treated; however, some subjective symptoms persist and these resolve over time without additional treatment. The family agreed to stop antibiotic therapy and over the next 6 weeks her arthralgias resolved.

My second story involved a 36-year-old female who was hospitalized because of fever and neutropenia. The patient had a 2-year history of arthralgias and multiple negative serologies for Lyme disease. The patient still believed she had Lyme disease and found a physician who did a B burgdorferi urine antigen test for Lyme disease that was positive. The physician diagnosed Lyme disease and initiated intravenous antibiotic therapy. On the 23rd day of cefotaxime therapy, the patient developed fever and neutropenia. She was hospitalized, the cefotaxime was stopped, and her fever and neutropenia resolved. Her arthralgias resolved over the next year without antibiotics. We could not find data supporting the use of B burgdorferi urine antigen test for the diagnosis of Lyme disease. We published this case¹¹ and later, the test was taken off the market. It now has been replaced by a second generation B burgdorferi urine antigen test, which also lacks published data supporting its use.

My last story involved a 4-year-old boy with arthralgias and myalgias and positive serology for B burgdorferi. During months of oral antibiotic therapy, his symptoms persisted. The parents insisted on intravenous ceftriaxone therapy, which was given reluctantly by a pediatric rheumatologist for 30 days. The patient's symptoms persisted. The parents then found another physician willing to continue the intravenous ceftriaxone therapy. On his eighth week of intravenous therapy, the patient developed acute cholecystitis and required a cholecystectomy. Additional antibiotic therapy was not given and the patient's symptoms resolved over the next few weeks.

I ended my presentation by reading from an article from the New York Times.¹² This article appeared on the front page and was entitled "Prolonged Lyme Treatments Posing Risks, Experts Warn." "Nearly 2 decades after Lyme disease was detected in Old Lyme, Connecticut, health experts and officials are warning that over-diagnosis of the disease and complications from long-term antibiotic treatments may pose as great a danger to public health as the disease itself. In 1 instance cited by the Federal Centers for Disease Control and Prevention, 14 children had to have their gallbladders removed, and 23 suffered blood stream infections because of complications of long-term treatment with intravenous antibiotics at the Jersey Shore Medical Center. Although the children's doctor defends their treatment, the center found no evidence that most of the children had Lyme disease."¹²

At the end of my presentation, I was asked by the Connecticut policymakersif a physician diagnoses Lyme disease and wants to treat the patient intravenously, if the risks of intravenous therapy are explained to the patient and/or family and they want intravenous therapy, then should the insurance company be able to interfere with this therapy? I answered that a plan agreed on between a physician and patient should not depend on a third party like an insurance company. However, the insurance company should not have to pay for tests or therapy that are not scientifically supported.

CONCLUSIONS

The following is a quote from Hartford's major newspaper, Hartford Courant¹⁴ describing the public hearing: "... what followed was a clash between 2 groups: patients and physicians who believe that insurers must be made to pay for long-term antibiotics if they seem to help; and insurers and researchers wary of the dangers and cost of over-treatment. Judging from applause and groans, the roughly 200-member audience was weighted heavily in favor of patients and family members who want insurers to pay for long-term care."¹⁴

In June 1999, the Connecticut legislature passed Public Act 99-2, which mandates health insurance companies to pay for not <30 days of intravenous antibiotic therapy for children and adults with presumed Lyme disease. Objective findings and/or positive B burgdorferi serology are not required. In addition, insurance companies are mandated to pay for >30 days of intravenous antibiotic therapy ordered in consultation with a board-certified rheumatologist, neurologist, or infectious disease specialist. Again objective findings and/or positive B burgdorferi serology is not required.

A public hearing is not a scientific forum. A physician who provides expert testimony at a public hearing can be best prepared by knowing the purpose and intent of their testimony and the audience that will be addressed. Patient advocacy groups may be influenced more by individual stories (testimonials) than by scientific studies. Lastly, the public may view nonacademic physicians, who reject standard diagnostic and treatment protocols, as being as authoritative as medical-center-based scientists.

There are 2 points that I would like to emphasize.¹ There are guidelines for the diganosis and treatment of Lyme disease,^5,6,7,14 and when these guidelines are followed, outcomes are excellent.^15-18 However, patient advocacy groups and a network of physicans believe that these guidelines are flawed and have begun what the lay press calls another Lyme camp.² Traditional medicine has failed the chronic Lyme disease patients who spoke at the Hartford public hearing. They were sincere in their testimony and believe that their symptoms improved with intravenous antibiotic therapy. Standard diagnostic and treatment protocols have not helped these patients. This testimony needs to be put into perspective by prospective, blinded, controlled studies. However, until such studies are completed, prolonged intravenous antibiotic therapy for patients with presumed Lyme disease should not be used.

Henry M. Feder and Jr, MD
Departments of Family Medicine and Pediatrics
University of Connecticut Health Center
Farmington, CT 06030-1406
Connecticut Children's Medical Center
Hartford, CT 06105

FOOTNOTES

Received for publication Jun 2, 1999; accepted Oct 29, 1999.

Address correspondence to Henry M. Feder, Jr, MD, Department of Family Medicine, MC 1406, University of Connecticut Health Center, 263 Farmington Ave, Farmington, CT 06030-1406. E-mail: feder{at}exchange.uchc.edu

REFERENCES

1. Feder HM Jr, Hunt MS Pitfalls in the diagnosis and treatment of Lyme disease in children. JAMA 1995; 274:66-68 [Abstract]
2. Reid MC, Schoen RT, Evans J, Rosenberg JC, Horwitz RI The consequences of overdiagnosis and overtreatment of Lyme disease: an observational study. Ann Intern Med 1998; 128:354-362 [Abstract/Free Full Text]
3. Barbour AG, Fish D The biological and social phenomenon of Lyme disease. Science 1993; 260:1610-1616 [Abstract/Free Full Text]
4. Burrascano JJ Jr. Lyme disease. In: Raker RE, ed. Conn's Current TherapyLatest Approved Methods of Treatment for the Practicing Physician. Philadelphia, PA: WB Saunders Co; 1997:140-143
5. Steere AC. Lyme disease. In: Raker RE, ed. Conn's Current TherapyLatest Approved Methods of Treatment for the Practicing Physician. Philadelphia, PA: WB Saunders Co; 1998:132-135
6. Centers for Disease Control and Prevention. Case definitions for public health surveillance. Morb Mortal Wkly Rep CDC Surveill Summ. 1990;39:19-21. RR-13
7. American Academy of Pediatrics, Committee on Infectious Diseases. Lyme Disease: 1997 Red Book. Elk Grove Village, IL: American Academy of Pediatrics; 1997:329-333
8. Amercian College of Rheumatology, Council of the Infectious Diseases Society of America Appropriateness of parenteral antibiotic therapy for patient with presumed Lyme disease. Ann Intern Med 1993; 119:513
9. Phillips SE, Mattman LH, Hulinska D, Moayad H A proposal for the reliable culture of Borrelia burgdorferi from patients with chronic Lyme disease, even those previously aggressively treated. Infection 1998; 26:364-367 [Medline]
10. Steere AC, Taylor E, McHugh GL, Logigian EL The overdiagnosis of Lyme disease. JAMA 1993; 269:1812-1816 [Abstract]
11. Feder HM Jr Testing in Lyme disease. JAMA 1990; 265:693
12. Schemo DJ. Prolonged Lyme treatments posing risks, experts warn. New York Times. January 4, 1994:Front page, A-1-B-5
13. Condon G. Differences are voiced over Lyme disease. Hartford Courant. February 26, 1999:A-2
14. Treatment of Lyme disease. Med Lett. 1997;39:47-48
15. Salazar JC, Gerber MA, Goff CW Long-term outcome of Lyme disease in children given early treatment. J Pediatr 1993; 122:591-593 [Medline]
16. Adams WV, Rose CD, Eppes SC, Klein JD Cognitive effects of Lyme disease in children. Pediatrics 1994; 94:185-189 [Abstract/Free Full Text]
17. Gerber MA, Shapiro ED, Barke GS, Parcells VJ, Bell GL Lyme disease in children in Southeastern Connecticut. N Engl J Med 1996; 335:1270-1274 [Abstract/Free Full Text]
18. Gerber MA, Zemel LS, Shapiro ED Lyme arthritis in children: clinical epidemiology and long-term outcomes. Pediatrics 1998; 102:905-908 [Abstract/Free Full Text]