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Ann Thorac Surg 2000;69:930-931
© 2000 The Society of Thoracic Surgeons


Case Reports

Lemierre’s syndrome with bilateral empyema thoracis

Marco Alifano, MDa, Nicolas Venissac, MDa, Françoise Guillot, MDa, Jérôme Mouroux, MDa

a Service de Chirurgie Thoracique, CHU de Nice, Hôpital Pasteur, Nice, France

Address reprint requests to Dr Mouroux, Service de Chirurgie Thoracique, Hôpital Pasteur, 30, Av de la Voie Romaine - BP 69, 06002 Nice Cedex 1, France


    Abstract
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A 31-year-old patient is described with thrombophlebitis of the right jugular vein, and anerobic septicemia (Lemierre’s syndrome). Multiple pulmonary abscesses and bilateral fibrinopurulent empyema were also present. Treatment included intravenous antibiotics, heparin, and video-assisted thoracoscopic debridement of pleural cavities. A favorable outcome was observed.


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Lemierre’s syndrome is an extremely rare life-threatening septic thrombophlebitis of a jugular vein with anerobic septicemia [1]. We report the case of a patient who developed a Lemierre’s syndrome with multiple pulmonary abscesses and bilateral fibrinopurulent empyema.

A 31-year-old white woman was admitted to the infectious disease department of our hospital for fever, mild dyspnea, and right cervical pain.

Her past medical history was remarkable for deep venous thrombosis (2 years previously), and a duodenal ulcer (1 year previously). Tobacco exposure (15 packs per year) was also present. In the 6 days before admission, she presented right-sided sore throat and fever spikes (to 38.5° C), but did not seek medical advice.

On physical examination she was pyretic (39° C) and tachycardic (heart rate 110 beats/min), but normotensive (120/80 mm Hg). No respiratory distress was present. There was good dental hygiene. There was tenderness and swelling along the course of the right internal jugular vein, and the covering skin was warm and moist. Right-sided cervical adenomegaly was also appreciable. Lung examination revealed crepitation in both middle thoracic areas. Abdominal examination revealed splenomegaly. Neurologic and osteoarticular examination disclosed no abnormality.

Laboratory examinations displayed leucocytosis (29 x 109/L) and increased erythrocyte sedimentation rate. Blood gas levels were as follows: PO2, 66 mm Hg; PCO2, 34 mm Hg; pH, 7.34. Serologic tests for the human immunodeficiency virus were negative. Fusobacterium necrophorum was grown from blood samples, and in vitro tests disclosed complete susceptibility to both penicillin G and metronidazole. Chest roentgenogram and thoracic computed tomographic scan revealed the presence of multiple bilateral cavitary lesions. Color Doppler ultrasonography of the neck displayed thrombophlebitis of the right jugular vein, and no evident collection was present.

Based on these data, diagnosis of Lemierre’s syndrome was established, and medical treatment was started immediately (penicillin G, 20.000.000 U/24 hours; metronidazole, 1.5 g/24 hours; intravenous heparin, 370 mg/24 hours). In spite of this, in the 3 days following, the dyspnea worsened and clinical conditions continued to deteriorate. A computed tomographic scan, performed on the fourth day of hospitalization, revealed the presence of bilateral multiloculated pleural effusion. Right thoracenthesis revealed the presence of frank pus in the pleural cavity. Both heparin and antibiotics were continued according to the same schedule, and the patient was referred to the thoracic surgery department.

Bilateral video-assisted thoracoscopy was performed under general anesthesia, with double-lumen intubation to permit single-lung ventilation. Three ports for each side were employed. Multiloculated effusions and all fibrinopurulent layers were evacuated under direct vision, and the lungs were completely freed from adherences with parietal pleura. No fibrotic peel covering lung surface was identified. Pleural cavities were abundantly irrigated, and two chest tubes (28F) per side were left in place. In the postoperative period, clinical conditions gradually improved. Fever resolved on the third postoperative day, and all thoracic drains were removed by the sixth postoperative day. Signs of septic jugular thrombophlebitis progressively improved, and were completely resolved on the seventh postoperative day. The patient was discharged on the tenth postoperative day. Penicillin and metronidazole were continued for 4 weeks, and heparin for 2 weeks.

At 1-year follow-up the patient is in excellent general condition, and her chest roentgenogram is completely normal. A definitive thrombosis of the right jugular vein is evident at color Doppler echography.


    Comment
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In the early 1900s, some authors reported the occurrence of anerobic septicemia caused by thrombophlebitis of the internal jugular vein [1]. In 1936, Lemierre gave a remarkably vivid description of the syndrome, pointing out the strict relationship with a recent episode of pharyngitis, and the frequent occurrence of septic pulmonary infarcts and suppurative arthritis [1]. In the preantibiotic era, the syndrome had a very high mortality rate [1]. With the systematic treatment of upper respiratory tract bacterial infections, the syndrome became exceptional, and the rare case reported was caused by untreated pharyngeal, tonsillar, or orodental infections [2, 3].

Fusobacterium necrophorum remains the most common pathogen associated with the syndrome. Its endotoxin seems to be the cause of the venous thrombosis [4]. Intravenous administration of high-dose antibiotics with anaerobic activity is mandatory [2], and the combination penicillin-metronidazole is frequently employed [2, 3]. Clinical improvement after the onset of antibiotic therapy is frequently slow, and in some cases the appearance of new distant abscesses is observed in the first days of antibiotic therapy [5]. According to some authors, such an event should require the change of the antibiotic regimen [2]; however, in the experience of others, complete recovery was achieved with the same antibiotic treatment, after a short period of apparently unfavorable evolution [5]. In our case, the antibiotic regimen was not changed, in spite of the appearance of multiloculated pleural effusion, over the course of the treatment. After the operation, pulmonary lesions cleared and clinical signs of septic thrombophlebitis resolved. The decision of continuing the same antibiotic treatment was also based on results of in vitro sensitivity tests.

Pleural effusion is possible in the course of Lemierre’s syndrome. Our patient presented with bilateral empyema, a veritably exceptional condition. At both sides, the empyema was in fibrinopurulent phase. There are no standard recommendations concerning the treatment of empyema at this stage, and it was pointed out that delay in adequate treatment has resulted, even recently, in as much as a 22% mortality rate [6]. According to the majority of authors, thoracotomy should be employed only in cases requiring lung decortication for the presence of a fibrotic peel, whereas video-assisted thoracoscopic debridement, or intrapleural fibrinolytic agents through a chest tube, represent the possible alternatives in the fibrinopurulent phase [7]. However, in a recently published randomized trial, it was found that a primary treatment strategy with thoracoscopic debridement, is associated with higher efficacy and shorter hospital duration, than a treatment strategy that utilizes chest tube-directed fibrinolytic therapy [8]. The rapidly deteriorating clinical conditions of our patient prompted us to chose the thoracoscopic approach, in order to achieve an immediate complete drainage of both pleural cavities. In fact, exhaustive debridement of pleural cavities and complete lung reexpansion were immediately possible.

Our report confirms that in order to prevent catastrophic events, timely medical and surgical treatment are mandatory in Lemierre’s syndrome. Thoracoscopic debridement of fibrinopurulent empyema should be considered a valid therapeutic option.


    References
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 Abstract
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 Comment
 References
 

  1. Lemierre A. On certain septicaemias due to anaerobic organisms. Lancet 1936;1:701-703.
  2. Venkataraman M.T., Policar M. Fever, sore throat, and pulmonary infiltrates in a 20-year-old man. Chest 1997;112:268-270.[Free Full Text]
  3. Mitre R.J., Rotheran E.B., Jr Anaerobic septicemia from thrombophlebitis of the internal jugular vein. Successful treatment with metronidazole. JAMA 1974;230:1168-1169.[Abstract/Free Full Text]
  4. Langworth B.F. Fusobacterium necrophorum. Bacteriol Rev 1977;41:373-390.[Free Full Text]
  5. Faussat J.M., Coste A., Roger G., Page B., Marrek H., Roulleau P. Oropharyngeal origin of septic thrombophlebitis of the internal jugular vein. Apropos of 3 cases. Ann Otolaryngol Chir Cervicofac 1993;110:445-449.[Medline]
  6. Kelly J.W., Morris M.J. Empyema thoracis. South Med J 1994;87:1103-1110.[Medline]
  7. Silen M.L., Naunheim K.S. Thoracoscopic approach to the management of empyema thoracis. Indications and results. Chest Surg Clin North Am 1996;6:491-499.[Medline]
  8. Wait M.A., Sharma S., Hohn J., Dal Nogare A. A randomized trial of empyema therapy. Chest 1997;111:1548-1551.[Abstract/Free Full Text]
Accepted for publication July 6, 1999.




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