Hepatitis-associated aplastic anemia and acute parvovirus B19 infection: a report of two cases and a review of the literature.

Hepatitis-associated aplastic anemia is rare in general, but occurs in up to 28% of patients receiving liver transplantation for fulminant non-A, non-B hepatitis. Cases are commonly young men with mild hepatitis but severe aplastic anemia. Although cases have been reported in association with hepatitis A, B, and C, most appear to be due to a non-A-B-C virus. We report two cases of acute hepatitis subsequently complicated by marrow hypoplasia in patients with acute parvovirus B19 infection. Hepatic manifestations of parvovirus B19 infection range from liver chemistry abnormalities to fulminant hepatic failure and aplastic anemia. Our cases demonstrate a less severe form of hepatitis-associated aplastic anemia, and together with other data, suggest that parvovirus B19 is at least one cause of hepatitis-associated aplastic anemia, and may be a heretofore underrecognized hepatotrophic virus.

Pilot study of antithymocyte globulin in systemic sclerosis.

OBJECTIVE: Between June 1, 1992 and August 31, 1994 we conducted an open pilot study of antithymocyte globulin (ATGAM; Upjohn, Kalamazoo, MI) in 10 patients with early systemic sclerosis (SSc) to assess whether this agent might prevent the progression of cutaneous and pulmonary involvement in this disease. METHODS: Adult patients with early SSc (< 3 years) and evidence of progressive skin and pulmonary disease were enrolled. All patients were hospitalized and received a single course of intravenous ATGAM, at a dosage of 10 mg/kg over 4 hours, on 5 consecutive days. Patients were followed up at weeks 1, 2, 3, and 4, and months 2, 4, 6, and 12. Patients were considered to be improved if the Rodnan skin score decreased > or = 25%, to be worse if the skin score increased > or = 25%, and to be not improved if the skin score was within 25% of baseline. For pulmonary involvement, patients were considered to be improved if either the diffusing capacity for carbon monoxide or the forced vital capacity was increased > or = 10%, worse if decreased by > or = 10%, and stable if within 10% of baseline. RESULTS: Most patients tolerated the treatment well, although 1 patient developed an allergic reaction necessitating discontinuation of treatment, 1 developed a serum sickness reaction after completion of therapy, and 1 developed a central venous access-related axillary vein thrombosis. Two patients died of SSc-related complications during the followup period. At 12 months, only 2 patients showed improvement in both skin and pulmonary function measures, whereas 5 patients were worse and 3 were stable. CONCLUSION: At the dosage administered in this study, ATGAM appears ineffective in improving the skin and pulmonary features of SSc.

Vasculitis associated with malignancy.

A large variety of vasculopathic syndromes are uncommonly associated with malignancies. Vasculitis is usually manifested by skin lesions and is generally associated with hematologic malignancies rather than solid tumors. Evidence of autoantibodies, immune complexes, and complement consumption is typically absent. Myelodysplastic syndromes can be confidently linked to vasculitis on the basis of recent literature. The temporal relationship of malignancy to vasculitis development is variable except that vasculitis generally follows the discovery of hairy cell leukemia and splenectomy. Vasculitis may occasionally be a complication of chemotherapy, radiation therapy, and bone marrow transplantation. Occasionally, malignant disorders may mimic vasculitic syndromes. The etiopathogenesis of vasculitis in patients with malignant disorders is unknown. The recent literature on vasculitis and malignancy addresses predominantly case reports and small patient cohorts and identifies clinical characteristics rather than pathogenic mechanisms.

Diagnosing Lyme disease: the contribution of serologic testing.

Lyme disease is a multisystem disorder caused by a tick-transmitted spirochete, Borrelia burgdorferi. The diagnosis is based on clinical findings in most patients, particularly those with erythema migrans or exposure to geographic locations endemic for the disease. Detection of a specific antibody to B. burgdorferi is a useful confirmatory test in many patients. In atypical cases, however, a positive test result can be pivotal for determining the diagnosis and can lead to institution of definitive treatment. Serologic testing should not be used indiscriminately to diagnose Lyme disease or as the sole basis for administration of antibiotic therapy.

Localized scleroderma (morphea) and antibody to Borrelia burgdorferi.

Enzyme-linked immunosorbent assay (ELISA) and immunofluorescence assay (IFA) were performed in 25 and 32 cases of morphea, respectively. The more sensitive and specific ELISA was positive in only 1 of 25 cases and the mean value was lower in cases of morphea than in controls. IFA showed minimally reactive titers in 6 (19%) of 32 cases. There were an additional six cases with borderline titers. These data indicate that there is no specific association between Borrelia burgdorferi infection and morphea but that patients with morphea tend to have circulating antibodies that are cross-reactive.

Focal encephalitis in a young woman 6 years after the onset of Lyme disease: tertiary Lyme disease?

A 19-year-old woman had severe focal inflammatory encephalitis. Six years previously, she had had classic untreated Lyme disease characterized by erythema chronicum migrans, bilateral facial palsies, and lymphocytic meningitis. During her recent encephalitic illness, Lyme disease serologic tests were positive by indirect immunofluorescence microscopy, enzyme-linked immunosorbent assay, and western blot (immunoblot technique) testing. We hypothesize that the patient's focal inflammatory encephalitis was a result of a persistent spirochetal infection of the central nervous system.