Introduction: Lemierre's syndrome, also known as necrobacillosis and postanginal septicemia had been described in multiple case reports in the early 1900's, but not until a comprehensive review in 1936 by Lemierre did the syndrome receive its name. Since the advent of antibiotics, case reports have become so infrequent that this syndrome rarely is recognized.
Case Presentation: A 22 year-old previously healthy male was referred to the University of North Carolina at Chapel Hill for evaluation of a new large left pleural effusion. Approximately two and one-half weeks prior to his presentation, he described the acute onset of an exudative pharyngitis. He had been given a prescription for erythromycin to treat a presumed Streptococcal pharyngitis, but vomited after the first dose and subsequently did not complete the course of therapy. Several days after the onset of the pharyngitis, he developed severe vomiting and diarrhea. A syncopal episode prompted an emergency room visit where he was given intravenous fluids and sent home. Vomiting and diarrhea continued with the subsequent development of fevers to 103 F and a productive cough. He returned to his local emergency room four days after his initial emergency room visit. Upon presentation, his temperature was 101.9 F, heart rate 150, respiratory rate 24 with a blood pressure of 111/64. Laboratory data revealed a WBC 7.1 with 53% segmented neutrophils and 36% band forms. Hemoglobin was 13.6, hematocrit 39.7 and platelet count 69,000. Chem 7 was most notable for a BUN of 29, creatinine of 1.4 and bicarbonate of 27. Urinalysis revealed a specific gravity of 1.020, pH 5.0, 2+ protein, 8 mg urobilinogen, trace leukocyte esterase, nitrite positive, 5-9 WBC's, 25-49 RBC's and bacteria. Stool analysis revealed rare WBC's and RBC's. He was admitted to a local hospital with diagnoses of dehydration, prostatitis, gastroenteritis, urinary tract infection, and thrombocytopenia. He remained hospitalized for three days receiving intravenous fluids and antibiotics. Blood cultures obtained upon presentation grew a gram negative rod. He was discharged to home to complete a seven day course of oral ciprofloxacin but subsequently developed severe left sided pleuritic chest pain, dyspnea and profound weakness in addition to continued high fevers, rigors and nausea with vomiting. Reevaluation by his local MD prompted a chest radiograph revealing a large left-sided pleural effusion. Repeat CBC also was notable for a WBC of 32,000. He was then referred to our institution for further evaluation. Upon presentation, he appeared toxic. His temperature was 39.3 C, HR 120, BP 120/72 with orthostasis, respiratory rate 28 and shallow with room air oxygen saturation of 90%. Physical exam revealed a resting tachycardia without murmurs or rubs, and signs of a large left-sided pleural effusion. Head, neck and oral exams were unremarkable. He was admitted and a diagnostic thoracentesis revealed an empyema (pH 6.58, glucose 20, protein 5.6, LDH 21,863, 1633 BBC, 14,933 WBC with 98% neutrophils and 2% lymphs). Gram stain of the pleural fluid revealed gram negative rods which were unable to be cultured. The gram negative rod in the blood cultures from the outside institution were identified as Fusobacterium necrophorum. He underwent chest tube thoracostomy in addition to receiving intravenous clindamycin and cefotaxime. Intrapleural fibrinolytic therapy was required for complete drainage of the empyema. His clinical status slowly improved, and he was discharged after two weeks. Further evaluations included a negative transesophageal ECHO and negative neck CT. Chest CT revealed a large left sided empyema and multiple right and left sided fluid collections but no evidence of mediastinitis. The patient was seronegative for HIV antibodies.
Discussion: Lemierre's syndrome is an acute oropharyngeal infection caused by anaerobic gram negative organisms, most commonly Fusobacterium necrophorum with secondary thrombophlebitis of the internal jugular vein and subsequent metastatic infection. This case illustrates several important features of Lemierre's syndrome. Affected individual are young, between the ages of 18 and 29 years old and are healthy with no comorbid diseases. Initial symptoms most commonly are that of a tonsillitis or pharyngitis, with or without an exudate. Tonsillar, peritonsillar or retropharyngeal abscesses may be present. Occasional presentations include otitis media, ethmoid or sphenoid sinusitis and odontogenic infections, but lung involvement is almost invariable. After presentation there may be transient improvement followed within 7 to 15 days by septicemia. High fever, tachycardia and respiratory distress are common later in the course of the disease but severe hypotension and shock are not manifest until the patient is near death. Physical findings may reveal unilateral or bilateral adenopathy in the anterior triangle of the neck and well as a palpable thrombosed internal jugular vein. Evidence of pulmonary involvement is frequent, ranging from pneumonia to pleural effusions. Cavitation of pulmonary lesions has not been remarked upon. Metastatic infections are common and include septic arthritis, soft tissue abscesses, cellulitis, meningitis, osteomyelitis, liver abscesses and endocarditis. Laboratory data commonly reveal thrombocytopenia and leukocytosis. The WBC may be in the normal range, however, with evidence of a left shift. Subclinical or overt hyperbilirubinemia as well as mild to moderate hepatic enzyme abnormalities are common. The organism is frequently isolated from either blood or other infected sites in anaerobic cultures. The diagnosis is made clinically, although computed tomography and doppler ultrasound of the neck can support the diagnosis. Treatment requires antibiotics with good anaerobic coverage such as penicillin G, clindamycin, metronidazole, cefoxitin and chloramphenicol as well as drainage of purulent collections. The role of anticoagulation is controversial.
Conclusion: Lemierre's syndrome can be easily diagnosed clinically because of its classical presentation. It is, however, infrequently recognized in the antibiotic era.
Jack Gilbey, MD and P. Bromberg, MD--University of North Carolina at Chapel Hill, Chapel Hill, North Carolina
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