Spirochetes in the Spleen of a Patient with chronic Lyme Disease
Marco A. Commino, M.D. ,Alfonso Azzolini, M.D. Franca Tobia, Dr. Sch. Biol.en Carlo M. Perce, M.D
Instituto Scientifico di Medicine Interna, Instituto di Anatomia Patologica, Université di Genova
Genova, Italy
1989
http://ajcp.oxfordjournals.org/content/ ... 5.full.pdf
Het middelste gedeelte heb ik weg gelaten want dat gaat vnl. over de soorten testen die er werden gedaan, en daar kwam niet veel bijzonders uit.
A 54-year-old man had intermittent evening fever, arthralgia, transient erythematous macular eruption on the skin, and splenomegaly of two year's duration. Immunofluorescence tests for Borrelia burgdorferi serum antibodies had positive results, but G-penicillin treatment was ineffective. Splenectomy with lymph node biopsy was performed to rule out lymphoproliferative disorders. Borrelia-like spirochetes were identified histologically in the spleen; this finding was consistent with persistence of B. burgdorferi organisms in inner organs in chronic Lyme disease
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Circulating immune complexes determined with an immunoenzymatic method showed a coefiicient of inhibition of 7% (nv less than 17%); no cryoglobulins were identified. There was a monoclonal light X chain spike in the serum (total IgG was 19.6 g/L [1.96 g/dL]; nv less than 17 g/L [1.7 g/dL]). A bone marrow smear showed hyperplasia of the myeloid cells and many eosinophils. Splenomegaly and enlarged paraaortic lymph nodes were seen with abdominal computer tomography. A skin biopsy showed nonspecific vasculitis, with perivascular cuffs and moderate diffuse infiltrates of lymphocytes with few plasma cells. IgG titers against B. burgdorferi antigen were determined by indirect immunofluorescence against B31-strain B. burgdorferi at the Istituto di Microbiologia, University of Ban. The patient's serum was absorbed with Treponema phagedenis according to the method of Wilske and colleagues18 to avoid cross-reaction with T. pallidum. The threshold level was set at 1:64 because none of 40 healthy blood donors had titers higher than 1:32. Three subsequent immunofluorescence tests yielded titers of 1:128. A VDRL test had negative results.
The patient was treated with G-penicillin (20 million units a day) intravenously. Transient reaction to treatment, which resulted in increased arthralgia and fever, appeared on the third day. After three weeks, treatment was considered ineffective and discontinued because fever continued and anemia progressed. Splenectomy and deep lymph node biopsies were performed to rule out lymphoproliferative diseases. The spleen weighed 550 g.
Histologically, only red-pulp hyperplasia was seen; a Warthin-Starry silver stain showed sparse Borrelia-like spirochetes (Fig. 1). No spirochetes were detected in the lymph nodes or in a liver biopsy specimen with normal histologic features. Another cycle of antibiotic treatment was suggested, but the patient, who had been discharged after splenectomy, refused readmission. Later the patient was treated with a second cycle of intravenous penicillin at another Institution, showing mild clinical improvement. Recently he was given a single intravenous dose of ceftriaxone (1 g); this treatment was discontinued because it elicited an allergic reaction. At present (April 1988) the patient is being treated with a regimen of prednisone (20 mg a day) and claims to be in good physical condition
Discussion
We believe that this patient was affected by an atypical form of LD with misleading clinical features. The three immunofluorescence tests suggested B. burgdorferi infection, which was later supported by identifying Borrelialike spirochetes in the spleen. The east Ligurian coast is an area endemic for LD, with more than 20 cases recognized since 1983.4 However, the patient did not experience any tick bite, and the onset of the disease was characterized only by fever, arthralgia, and lymphosplenomegaly. The transient, nonpruriginous macular eruption corresponding histologically to nonspecific lymphocytic vasculitis might have represented a variant of erythema chronicum migrans.3 Finally, failure of penicillin treatment is not in contrast to the diagnosis of LD because
this antibiotic is known to be occasionally ineffective in B. burgdorferi infections.15 B. burgdorferi has been identified in the blood, cerebrospinal fluid, skin,16 myocardium,7 kidney,9 eye,14 synovial fluid,12 and synovium6 of patients affected by LD. Splenomegaly is found in 6-8% of them1 u3 and has been ascribed to direct invasion of the spleen or to infectionrelated toxic reactions. Pathologic data concerning spleen involvement in LD are scanty, however.