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* From the Departments of Pediatrics (Drs. Bowsher and Callahan), Clinical Investigation (Dr. Person), and Radiology (Dr. Reuss), Tripler Army Medical Center, Honolulu, HI.
Correspondence to: Charles W. Callahan, DO, LTC, MC, USA, FCCP, Tripler Army Medical Center (MCHK-PE), 1 Jarrett White Rd, Honolulu, HI 96859-5000
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Abstract |
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Key Words: anti-I cold hemagglutinin • leptospirosis • pneumonia
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Introduction |
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Case Report |
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A repeat chest radiograph was unchanged, but CT of the chest revealed a large right sided pleural effusion with a mild left mediastinal shift. A chest tube was inserted, draining 400 mL of hazy, yellow exudative fluid that clotted (total protein, 3.8 g/dL; lactic dehydrogenase, 2,525 U/L; and specific gravity, 1.020). At this point, her antibiotic therapy was changed to oxacillin and gentamicin, and she was emergency evacuated to Tripler Army Medical Center (TAMC).
Upon arrival at TAMC, her heart rate was 144 beats/min; respiratory rate, 50 breaths/min; BP, 111/40 mm Hg; and temperature, 37.2°C. She was alert and oriented. Her sclerae were icteric, and conjunctivae were injected. A right sided chest tube was in place, and auscultation revealed fine crackles on the right with clear breath sounds on the left. A cardiovascular examination was significant for tachycardia. An abdominal examination revealed hepatomegaly 2 cm below the right costal margin, without splenomegaly. Her skin was jaundiced.
A laboratory evaluation revealed a WBC count of 15.6 x 103/µL; hemoglobin, 8.4 g/dL; hematocrit, 24.5%; and platelets, 33 x 103/µL. On urinalysis, RBCs were 27/high power field (hpf). Her prothrombin time was 13.0 s (normal), and partial thromboplastin time was 27 s (normal). Her aspartate aminotransferase level was elevated at 185 U/L. Her amylase was 165 U/L (normal), and lipase was elevated at 1,183 U/L. Serum albumin was 2.1 g/dL, and total bilirubin was 5.1 mg/dL. Creatinine was 0.9 mg/dL, and her BUN was 21 mg/dL. The pleural fluid analysis was repeated, revealing a pH of 7.5; glucose, 21 mg/dL; total protein, 4.5 g/dL; lactic dehydrogenase, 17,310 U/L; WBC, 800/hpf; and RBC, 1,790/hpf. The result of a qualitative test for cold agglutinins performed at bedside was positive. (Two milliliters of whole blood were placed into a blue-top sodium citrate tube and mixed. The side of the tube was noted to have a uniform coat of blood against a white background. The tube was then placed on ice for 3 to 5 min, and removed. When held against a white background again, the granular appearance of cold agglutinated blood was noted. The agglutination resolved when the tube was rewarmed.)
Her chest radiograph revealed right upper and middle lobe airspace
disease with pleural effusion (Fig 1
). Repeat CT of the chest revealed dense consolidation of most of the
right lung with air bronchograms and pleural effusion (Fig 2
, top and bottom). Treatment with nafcillin,
cefotaxime, and erythromycin was begun. Over the next 36 h,
her peripheral smear showed signs of hemolysis. Her hemoglobin dropped
to 6 g/dL; hematocrit, 15.8%; red (blood cell) distribution
width, 15.4% (high); and total bilirubin rose to 23.3 mg/dL
(unconjugated, 1.6 mg/dL; conjugated, 15.8 mg/dL; and 
bilirubin,
5.8 mg/dL). Her BUN and creatinine peaked at 37 mg/dL and 0.9
mg/dL, respectively. Her prothrombin time climbed to 15.9 s. Her
aspartate aminotransferase was elevated at 200 U/L, and 
-glutamyl
transpeptidase rose to 152 U/L. The hemolysis was found to be due to an
anti-I antibody (not quantified). Her bone marrow aspirate and biopsy
specimen were interpreted as hyperplastic. Because her clinical picture
was evolving to be more consistent with leptospirosis, penicillin was
added to the antibiotic regimen. Her autoimmune hemolysis was treated
with IV "pulse" doses of high-dose methylprednisolone, 30 mg/kg
every 8 hours, resulting in a dramatic increase in platelet count, a
resolution of hemolysis, and clearance of the pneumonic process.
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The results of serial mycoplasma antibodies were negative. The results of all blood cultures and cultures of the pleural effusion were negative. The result of a urine culture for Leptospira was negative. She completed 1 week of treatment with nafcillin and cefotaxime, and 10 days of treatment with penicillin and erythromycin. Her radiographic findings improved, and by 2 weeks she had completely recovered.
Serum samples obtained at 1 and 2 weeks after the onset of her illness were negative for leptospiral antibodies, both by direct hemagglutination (State of Hawaii Department of Health, Honolulu, HI) and microscopic agglutination (Centers for Disease Control and Prevention, Atlanta, GA.) A third serum sample (obtained 23 days after the onset of symptoms) tested positive by mircoscopic agglutination (Centers for Disease Control and Prevention) at a titer of 1:100 for Leptospira interogans serovar Bratislava, strain Jez-bratislava, a positive seroconversion. There were > 20 serovar/strain assays performed on each of the three sera, and all were negative except the above, which is diagnostic for leptospirosis.
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Discussion |
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Our patient had epidemiologic evidence to suggest leptospirosis, the clinical picture of Weil's disease, as well as positive leptospiral antibodies. She also had pancreatitis, a recognized complication of leptospirosis in children.1 2 3 5 Her serum leptospira titers were positive, and L interogans serovar Bratislava is a well-known cause of porcine-associated leptospirosis in the Pacific Basin. Her primary problem, however, was pulmonary disease (specifically, unilateral pneumonia with pleural effusion).
Unilateral pneumonia with effusion is common in pediatrics. There are a variety of bacterial etiologic agents that may lead to this clinical picture, including Streptococcus pneumoniae, Staphylococcus aureus, and invasive Haemophilus influenza.6 Disease from Mycoplasma pneumoniae may also produce unilateral pneumonia with pleural effusion.7 In addition, M pneumoniae may also cause hemolytic anemia related to cold agglutinins (anti-I antibody), thrombocytopenia, mild hepatitis, pancreatitis, and GI symptoms.7 Our patient's initial course was typical for a complicated Mycoplasma infection, thus her early treatment included macrolide therapy. A lack of response to therapy, the severity of her associated symptoms, and the absence of measurable antibody titers to M pneumoniae on serial examinations suggested another etiology.
Infection due to leptospirosis is thought to cause a systemic vasculitis, with damage to the capillary endothelium being the main site of the vascular damage. Focal hemorrhage of the tracheobronchial tree, parenchyma, diaphragm, and pleural surfaces have been reported with pleural effusions that are exudative and occasionally hemorrhagic. Radiographic abnormalities are most commonly noted in the first week of the disease in 6 to 10% of adults, and they are usually due to scattered alveolar hemorrhage in a "snowflake-like" or "mottling spot" pattern, most frequently in the lower lobes.8 In a series of 58 patients with leptospirosis and lung disease, radiographic abnormalities were noted in 37 patients (64%). All had bilateral disease, seven had pleural effusion (19%), and in six patients it was right sided.9 In a recent series of 43 children with leptospirosis, 3 patients developed hemorrhagic pulmonary disease, and it was the cause of the single death in the series.10 Pneumonitis with pleural effusions, albeit much less severe than that reported here, has been observed in three of nine children hospitalized with leptospirosis at TAMC in the past 3 years.1 2 3
The clinical presentation of this patient was also complicated by a hemolytic anemia. Anemia and thrombocytopenia are commonly reported with leptospirosis. Hemolysis is less common, and the mechanism is not well understood. This contributed to the confusion in the initial diagnosis for our patient. We also report what we believe to be the first association of leptospirosis with hemolysis due to anti-I hemagglutinin disease. Cold hemolysis is induced by IgM antibodies that bind to the RBC I/I antigen system.11 Although cold hemagglutinin disease may be associated with infections due to Epstein-Barr virus, cytomegalovirus, and mumps, anti-I hemagglutinin is most commonly associated with M pneumoniae.7
Pneumonia that is unilateral with pleural effusion and associated with cold hemagglutinin disease due to anti-I IgM would lead almost any clinician to think of Mycoplasma infection and treat accordingly. However, leptospirosis is an infection that causes generalized vasculitis with a wide array of clinical manifestations. It should be suspected, tested for, and treated in endemic areas when the history suggests the appropriate risk factors for zoonotic infection.
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Footnotes |
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The opinions expressed are those of the authors and do not necessarily reflect the position or policy of the Department of Defense, the Department of the Army, or the Army Medical Department.
Received for publication September 29, 1998. Accepted for publication April 7, 1999.
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