Sociedade portuguesa de medicina interna - volume 1 #3 october/december 1994 - english version
Lyme disease: a rare form of presentation
P. Ramos*, C. Fernandes**, J. Moreira***, A. Ventura****, M. Riscado****
The authors present the case of a 32 year old male presenting
doxycycline, with rapid disappearance of al the symptoms. In the
classic features of Erythema Chronicum Migrans. Significant titers
fol owing months, at almost monthly intervals, the patient suffered
for Borrellia burgdorferi were then obtained. Following antibiotic
multiple episodes of recurrence with widespread secondary cuta-
treatment with tetracycline, a good response was obtained, with
neous lesions characteristic of Lyme disease. Definitive remission
rapid disappearance of all the skin lesions. After two months,
was then obtained with the use of ceftriaxone IV.
new secondary skin lesions of Lyme disease were observed, to-
Key words: Lyme disease, periorbital edema, purulent con-
gether with intense, bilateral and periorbital edema and purulent
conjunctivitis. Once again, a good response was obtained with
Migrans stage of Lyme Disease was subsequently
Lyme disease is an infection with varied clinical mani-
confirmed through positive serology with signifi-
festations and it is currently well defined. The ocular
cant titers against Borrelia burgdorferi (IgM:1/320).
compromise that is characteristic of the secondary
Therapy with tetracycline (500 mg. t.i.d., P.O.) was
phase of the disease is an infrequent form of manifes-
then administered for twelve days, with complete
tation, occurring in only around 3% of cases¹.
remission of the condition and a clear reduction in
Resistance to antibiotics, as well as certain clinical
serum titers in the subsequent weeks.
manifestations, appears to be associated with parti-
Approximately two months afterwards, the pro-
cular immunopathogenic characteristics, in relation to
gressive appearance was observed of multiple round
H.L.A², as we shall see in the case described here.
skin lesions, pruriginous, with a clearly-defined, round border, affecting both limbs, the trunk, face
and scalp, resulting in hospitalization.
A 32 year old male, white, a soldier, residing in
Twenty four hours after the observation, unex-
Amadora, healthy, examined in August 1992 due to
pected rapid, bilateral onset of intense periorbital
the appearance, about six weeks previously, of pru-
edema and purulent conjunctivitis was observed,
riginous red pimples on the left knee and trunk, of
with inability to open the eyelids, fever (37.9°C) and
about 10 cm in diameter, confluent, migratory, and
arthralgias, particularly involving the metacarpus-
developing in about 3-5 days (figures 1 & 2). The
phalangeal joints, elbows and knees.
patient had already been treated with ampicillin and
Subsequent tests carried out in the laboratory
amoxicillin at the outpatient clinic, without any clinical
showed unaltered hemogram, ESR 10 mm in the first
hour, biochemistry unaltered (including P.F.H., P.E.R.
A clinical suspicion of the Erythema Chronicum
and urine type II), coagulation within the normal li-mits, negative VDRL, immunoglobulin levels without alterations, CPR. = (4+), negative Widal, Huddlesson and Wright, cryoglobulin (2+), C.I.C. 7ug/ml, and HLA (+) for Drw2 and Drw4.
Given the evidence of multiple secondary impair-
*Resident to the Internal Medicine Supplementary Internship
ment (ocular, periorbital and cutaneous involvement)
**Resident to the Dermatology Supplementary Internship
characteristic of Lyme disease (stage II), tetracycline
therapy was initiated (500 mg, q.i.d, P.O. over 15
****Internal Medicine Hospital Senior Assistant
days), with excellent results; the symptomatology was
Medicine Service 2 and Dermatology Service.
completely reduced on the 5th day of therapy.
Over the nine subsequent months, seven episodes
PUBLICAÇÃO TRIMESTRAL VOL. 1 | Nº 3 | OUT/DEZ 1994 CASE REPORTS Medicina Interna
of recurrence of secondary cutaneous lesions were
of all cases (48%).³,4,7 These were also found in the
observed, with similar characteristics and evolution
case described, demonstrating the ineffectiveness
to those diagnosed previously, and which were not
of the treatment and progression of the disease to a
accompanied by any other symptomatology. The in-
further level. 6 Neurological and cardiac impairment
terval of respite between recurrences was, on average,
are also common (present in 15% and 8% of cases),
35 days (±4 days), on which the patient remained
characterized by meningeal irritation, peripheral fa-
asymptomatic. The serum titers remained high, with
cial paralysis, neuritis or cardiac blockage to varying
significant values (1/200, 1/156, 1/225), during the
degrees, respectively.5,7 Ocular impairment, manifes-
nine months in which recurrences were observed.
ted in this case by purulent bilateral conjunctivitis
The various episodes were treated in the same way,
and periorbital edema, is a rare form of presentation,
with tetracycline (500 mg q.i.d for 15 days) as well as
occurring in only 11% and 3% of cases.1,7,8
doxycycline (100mg b.i.d. for 15 days) and the symp-
The uncharacteristic evolution, marked by succes-
toms rapidly decreased after the start of treatment.
sive episodes of recurrence after therapy, and the rare
Given the clear evidence of resistance to the anti-
visceral compromise (ocular involvement) observed
biotic therapy, and following the appearance of new
may be related to the particular immunopathogenic
secondary lesions similar to those previously descri-
characteristics of the individual patient. Indeed, a
bed, ceftriaxone (3 gr/day/I.V. for 10 days) was pres-
particular association has been found between HLA-
cribed, under hospitalization, with rapid resolution
Drw2/Drw4 and the evolution to chronic disease,1,7,9
and remission of the symptoms. Since then there have
in which resistance to classic antibiotic treatment is
been no other recurrences, and after several months,
The therapy, which generally involves tetracycline
or doxycycline1,2, was clearly ineffective in this case.
Therefore, in apparently resistant forms, or those asso-
The case described above shows the unpredictable
ciated with a typical visceral compromise in advanced
variability classically associated with Lyme disease.
stage, intravenous use of penicillin or a 3rd generation
Initiating, in this case, with the characteristic cuta-
cephalosporin of (ceftriaxone) is prescribed.10
neous lesion of Erythema Chronicum Migrans and confirmed by serology in significant titer², it was not,
however, accompanied by the general manifestations
1. Steere C, Bartenhagen D et al. The Early Clinical Manifestations of Lyme
usually associated with it, such as fever, arthralgia,
2. Volkman D, Dattwyler R. Imunodiagnóstico e tratamento da Borreliose de Lyme. Momento Médico 1990; 30(3): 3-11.
The appearance of multiple round cutaneous le-
3. Steere A, Malawista S, Hardin J, Ruddy S, Askenase P, Andiman W. Erythema
sions is a frequent finding, occurring in around half
Chronicum Migrans and Lyme arthritis. Ann Intern Med 1977: 86: 685-698. REVISTA DA SOCIEDADE PORTUGUESA DE MEDICINA INTERNA CASE REPORTS Medicina Interna
4. Luger S. Lyme disease transmitted by a biting fly. Letter – N Engl J Med 1990; 322:1752. 5. Sigal L. Summary of the first 100 patients seen at a Lyme Disease Referral Centre. AM J Med 1990; 88: 577-581. 6. Steere A, Hutch G, Rahn D, Sigal L, Craft J, Desana E, Malawista S. Tre-atment of the early manifestations of Lyme disease. Ann Intern Med 1983; 99: 22-267. Ruel M, Dournon E. La Maladie de Lyme. Ann Med Interne 1988 ; 139 : 555-574. 8. Aaberg T. The expanding ophthalmologic spectrum of Lyme disease. Am J Ophth 1989; 107:77-80. 9. Steere A, Dwyer E, Winchester R. Association of chronic Lyme Arthritis with HLA-Dr4 and HLA-Dr2-alleles. N Engl J Med 1979; 301 (25): 1358-136310. Dattwyer R, Halpering J, Volkman D, Luft G. Treatment of late Lyme borreliosis – Randomized Comparison of ceftriaxone and penicillin. Lancet 1988, 8596: 1191-1194.
PUBLICAÇÃO TRIMESTRAL VOL. 1 | Nº 3 | OUT/DEZ 1994
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