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Radiological Case of the Month
Teresa Rivera-Penera, MD;
Karin Nielsen, MD, MPH;
Theodore R. Hall, MD
Arch Pediatr Adolesc Med. 1998;152:87-88.
DURING THE early fall, a previously healthy 10-year-old child had a fever (temperature, 39.4°C) and upper respiratory tract symptoms of cough and rhinorrhea. No abnormalities were seen on a chest radiograph. His fever persisted despite a 10-day course of clarithromycin. On reexamination the patient was found to have acute otitis media and sinusitis, which were treated with the combination drug, amoxicillin–clavulanate potassium for 10 days. He continued to have a low-grade fever, malaise, and exudative tonsillitis, and developed generalized lymphadenopathy, night sweats, and, abdominal, back, and flank pain 5 weeks after the onset of illness. Urine culture and Epstein-Barr Virus titers were negative. Abdominal radiographs showed no abnormalities, but an abdominal ultrasound examination showed cystic lesions in the liver. He was subsequently referred for further evaluation.
The patient had a history of upper respiratory tract infections. The patient's history also revealed that he had been exposed to many animals including chickens, turkeys, salamanders, dogs, cats, rats, tarantulas, and sheep. Both parents and 2 siblings were healthy except for a history of respiratory allergies.
On physical examination, the patient was noted to have hepatomegaly and generalized lymphadenopathy. Preliminary laboratory test results showed a total white blood cell count of 11.0x103 cells/mm3, a hemoglobin count of 130 g/L, a hematocrit of 0.37, and a platelet count of 289x109/L. The patient's liver enzyme, amylase, and electrolyte values were within the normal range. An abdominal computed tomographic scan (Figure 1), with contrast, showed high-attenuation nodular fluid–containing lesions of soft tissue density that were diffusely distributed in the liver. The largest lesion measured 2.2 cm in diameter. A computed tomographic scan of the sinuses showed pansinusitis. A chest computed tomographic scan showed irregular soft tissue densities in the posterior lung bases bilaterally representing areas of atelectasis. Multiple serologic examinations were obtained including those for human immunodeficiency virus, hepatitis A, B, and C, cytomegalovirus, Echinococcus sp, Entamoeba histolytica, Toxocara sp, Mycoplasma, Toxoplasma, and alpha1-fetoprotein. A single photon emission computed tomographic technetium Tc 99m sulfur colloid liver-spleen scan was performed to differentiate between focal nodular hyperplasia and hepatic adenomas (Figure 2). The procedure demonstrated multiple "cold" liver defects and splenomegaly. A single photon emission computed tomographic red blood cell liver scan tagged with 99m Tc microlite showed no hemangiomas.
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Figure 1.
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Figure 2.
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The patient continued to have a low-grade fever. He was treated for his sinusitis with intravenous cefotaxime sodium. On the third day in the hospital, a liver biopsy was performed. Necrotizing granulomas, extensive neutrophilic infiltrates, and plasma cells were present. Gram stains and special stains for acid-fast bacteria and fungus showed no microorganisms. A Steiner-Steiner stain for cat-scratch disease revealed elements highly suspicious, but not definite, for Bartonella henselae. Tissue specimens cultured for bacteria, acid-fast bacteria, and fungus were negative. Polymerase chain reaction on liver tissue revealed B henselae DNA. There was no evidence of Bartonella quintana DNA.
Serologic test results were all negative except for B henselae and titers of IgG of 110 optical deurity (OD) and IgM of 36 OD (cutoff 12 OD). Bacterial urine and blood cultures were negative for organisms. Purified protein derivative testing gave negative results with positive tetanus controls. An absolute CD4 cell count performed initially showed 0.43x109/L.
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Denouement and Discussion: Granulomatous Hepatitis in Cat-scratch Disease
Figure 1. A spiral computed tomographic scan of the abdomen shows 4 well-defined, hypodense, hepatic lesions (arrows) with peripheral contrast enhancement. There is also a large area of low attentuation in the posterior segment of the right lobe of the liver.
Figure 2. The single photon emission computed tomographic technetium Tc 99m sulfur colloid liver-spleen scan shows inhomogeneous uptake of tracer in the right lobe with focal areas (arrowheads) of absent tracer uptake corresponding to the spiral computed tomographic abnormalities.
Cat-scratch disease (CSD) is caused by an argyrophilic, gram-negative, pleomorphic bacillus known as Bartonella (Rochalimaea) henselae. The diagnosis is made when 3 of the 4 following criteria are satisfied: (1) history of cat contact, associated with skin or eye inoculation, (2) positive CSD skin test, (3) normal laboratory study findings for other causes of lymphadenopathy, and (4) characteristic histopathologic features in the biopsy specimen of the involved skin or lymph node.1
The disease typically presents a fever and regional lymphadenopathy located adjacent to the inoculated site within 2 weeks after exposure to the cat. The cat is usually less than 1 year of age. The inoculated site appears as a papule 3 to 5 days after exposure and becomes vesicular and crusty. The involved lymph nodes may remain enlarged for months. Axillary lymphadenopathy is common because the hands and arms are the usual sites of inoculation. Other sites include the cervical and the inguinal regions. The histopathologic feature of the involved lymph node is a granuloma with central necrosis, infiltrating neutrophils, and lymphoid hyperplasia. The course of the disease is self-limited and seldom needs medical therapy. About 10% to 12% of the patients have unusual presentations making the diagnosis less obvious.2 Granulomatous hepatitis presenting with fever and abdominal pain, with or without lymphadenopathy, has been reported in the past.3-4 Rare presentations of CSD include Parinaud oculoglandular syndrome, acute encephalopathy,5 osteolytic lesions,6 transverse myelitis,7 neuroretinitis, aseptic meningitis,8 thrombocytopenic purpura, atypical pneumonia, and erythema nodosum.2 Gastrointestinal manifestations of CSD may include granulomatous splenitis and appendicitis.9 Atypical presentations may mimic other serious illnesses, such as lymphoma, tuberculosis, and malignancies. If the diagnosis of CSD is not considered early on, unnecessary, extensive and costly workup may ensue.
Of particular interest in this case is the extensive radiologic workup of the suspicious hepatic hypoechoic lesions found on the ultrasound film. With the findings of the computed tomographic, liver-spleen, and tagged red blood cell scans, we were able to rule out cystic lesions, benign nodular hyperplasia and hemangioma, respectively. Hepatocellular adenoma was considered as a possibility due to the detection of multiple cold defects on the liver-spleen scan. An open liver biopsy was performed. Operative findings revealed multiple nodules and histology showed necrotizing granulomas with special staining suspicious for CSD. The liver tissue was positive by DNA polymerase chain reaction for B henselae. Enzyme-linked immunosorbent assay serologies for B henselae were also positive.
The disease is self-limiting and seldom needs antibiotic therapy. However, in some cases of extensive lymphadenopathy and liver-spleen involvement, intravenous gentamicin has been used with success.10-11 In another study, ciprofloxacin has been shown to be an effective therapy for CSD.12
AUTHOR INFORMATION
Accepted for publication December 16, 1996.
Reprints: Theodore Hall, MD, Radiological Science BL-428, CHS, 10833 Le Conte Ave, Los Angeles, CA 90095.
REFERENCES
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1. Schartzman WA. Infections due to Rochalimaea: the expanding clinical spectrum. Clin Infect Dis. 1992;15:893-902.
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2. Margileth MA, Wear DJ, English CK. Systemic cat-scratch disease: report of 23 patients with prolonged or recurrent severe bacterial infection. J Infect Dis. 1987;155:390-402.
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3. Malatack JJ, Jaffe R. Granulomatous hepatitis in three children due to cat-scratch disease without peripheral adenopathy. AJDC. 1993;147:949-953.
4. Dangman BC, Albanese BA, Kacica MA, Lepow ML, Wallach MT. Cat-scratch disease in two children presenting with fever of unknown origin: imaging features and association with a new causative agent, Rochalimaea henselae. Pediatrics. 1995:5:767-771.
5. Noah DL, Bresee JS, Gorenselc, et al. Cluster of five children with acute encephalopathy associated with cat-scratch disease in South Florida. Pediatr Infect Dis J. 1995;14:866-869.
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6. Carithers HA. Cat-scratch disease: an overview based on a study of 1,200 patients. AJDC. 1985;139:1124-1133.
7. Pickerill RG, Milder JE. Transverse myelitis associated with cat-scratch disease in an adult. JAMA. 1981;246:2840-2841.
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8. Wong MT, Dolan MJ, Lattuada CP, et al. Neuroretinitis, aseptic meningitis, and lymphadenitis associated with Bartonella henselae infection in immunocompetent patients and patients infected with HIV type 1. Clin Infect Dis. 1995;21:352-360.
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9. Delahoussaye PM, Osborne BM. Cat-scratch disease presenting as abdominal visceral granulomas. J Infect Dis. 1990;161:71-78.
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10. Bogue CW, Wise JD, Gray GF, Edwards KM. Antibiotic therapy for cat-scratch disease? JAMA. 1989;262:813-816.
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11. Musso D, Drancourt M, Raoult D. Lack of bactericidal effect of antibiotics except aminoglycosides on Bartonella henselae. J of Antimicrob Chemother. 1994;36:101-108.
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12. Holley P Jr. Successful treatment of cat-scratch disease with ciprofloxacin. JAMA. 1991;265:1563-1565.
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SECTION EDITOR: BEVERLY P. WOOD, MD
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