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American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 159 – 162 www.elsevier.com/locate/amjoto

Lemierre syndrome: a case report
U. Kisser, MDa,⁎, R. Gurkov, MDa , W. Flatz, MDb , A. Berghaus, MDa , O. Reichel, MDa
a

Department of Otorhinolaryngology, Head and Neck Surgery, University of Munich, Munich, Germany b Department of Clinical Radiology, University of Munich, Munich, Germany Received 1 December 2010

Abstract

Introduction: Lemierre syndrome, also known as postanginal sepsis, is caused by Fusobacterium necrophorum. This rare disease is usually characterized by thrombophlebitis of the jugular vein and septic embolism after a history of sore throat. Objective: Here, we discuss a case of Lemierre syndrome in a 22-year-old man with thrombophlebitis of the facial vein and fusobacteria growth in the blood culture but no obvious focus of inflammation. Method: Case report. Conclusion: Severe facial infection with high fever and a general feeling of malaise after a history of sore throat should raise the diagnostic possibility of facial vein thrombophlebitis due to F. necrophorum infection. © 2012 Elsevier Inc. All rights reserved.

1. Introduction Lemierre syndrome, also known as postanginal sepsis, is caused by the anaerobic Fusobacterium necrophorum. This rare disease is usually characterized by thrombophlebitis of the jugular vein and septic embolism after a history of sore throat. We discuss a case of Lemierre syndrome in a 22-yearold man with thrombophlebitis of the facial vein and fusobacteria growth in the blood culture but no obvious focus of inflammation. 2. Case A previously healthy 22-year-old man with no medical history was admitted to the department of otorhinolaryngology, head and neck surgery, of our tertiary referral center after a 4-day history of a sore throat and generalized feeling of malaise. On day 3 of his illness, he developed a

⁎ Corresponding author. Department of Otorhinolaryngology, Head and Neck Surgery, University of Munich, Marchioninistraße 15, 81377 Munich, Germany. Fax: +49 89 7095 3850. E-mail address: ulrich.kisser@med.uni-muenchen.de (U. Kisser). 0196-0709/$ – see front matter © 2012 Elsevier Inc. All rights reserved. doi:10.1016/j.amjoto.2010.12.008

progressive, tender right facial edema with cellulitis as well as fever of 40.3°C and shivering. Examination of the oral cavity showed no signs of tonsillitis, peritonsillar abscess, or sialadenitis. The facial skin, however, showed signs of a thrombophlebitis of the right facial and angular vein (Figs. 1 and 2). The blood pressure was 210/100 mm Hg; the heart rate was 130 beats per minute. Laboratory tests showed leukocytosis (14.1 G/l [4.0– 11.0]), an increased C-reactive protein serum concentration (14.9 mg/dL [b0.5]), an increased procalcitonin level (11.4 ng/mL [b0.1]), and an interleukin-6 concentration of 38.9 pg/mL (b5.9). Ultrasound of the neck showed an abscess-like lesion in the area of the right submandibular gland. With the suspected diagnosis of submandibular abscess and in light of the deteriorating general condition of the patient, it was decided to perform a surgical drainage. Simultaneously, intravenous antibiotic therapy with clindamycin was started. Intraoperatively, the submandibular gland appeared slightly inflamed, but the actual focus of inflammation turned out to be in the right facial vein, which was affected by thrombophlebitis. Yellowish liquid was found intravascularly and sent for microbiological evaluation and culture. The thrombophlebitis did not yet reach the jugulofacial vein junction. The thrombotic facial

160 U. 3. On day 10. and ibuprofen. Pulmonary embolism could be excluded. First. but there were no signs of thrombosis of the jugular vein. Facial swelling on presentation of the patient. which were eventually identified as belonging to the species of F.4 G/l. necrophorum by day 6. clindamycin. From day 3 onward. 4). or the intracranial sinuses. When the pathogenic agent was identified on day 6. the patient was treated with moxifloxacin. 2. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 159–162 Fig. 3). MRI scan of the head and neck region showing inflamed soft tissue (→) surrounding the occluded facial vein. when the C-reactive protein level reached 27. The scan also showed extended reactive cervical lymphadenopathy. but no inflammatory focus could be found in the respective fields. Kisser et al. twice daily). however. Contrast-enhanced CT scan of the head and neck showing the occluded facial vein on the right side (→) and a normal facial vein on the left (⁎→). Blood cultures grew Gram-negative anaerobic filamentous bacteria by day 2. extended facial vein thrombosis was found (Fig. Fig. enoxiparin (80 mg.6 mg/dL and the leukocyte concentration was 6. the C-reactive protein level was 0. Fig. .2 mg/dL and leukocytes rose to a concentration of 17.2 G/l. It took 6 days until the facial cellulitis and edema slowly regressed (Fig. the ophthalmic vein. an increase in inflammatory parameters could be observed by day 2. the inflammatory parameters continually dropped. On computed tomography (CT). 5 shows patient 3 weeks after hospital admission). On days 1 to 5. 1. but there were several infectious metastatic abscesses in both lungs (Fig. the antibiotic treatment was continued using clindamycin combined with meropenem upon the recommendations of our bacteriologists. vein was partially removed and sent to the pathology and the microbiology departments. The patient was seen by an ophthalmologist and by a dentist.

Lemierre himself already mentioned cases of facial vein involvement [5]. was only slightly enlarged because of edema. or peritonsillar abscess. The latter. in the depth of an abscess. In conclusion. Usually. severe facial infection with high fever and a general feeling of malaise after a history of sore throat should raise the diagnostic possibility of facial vein thrombophlebitis due to F. Patient after treatment. The causal lesion of Lemierre syndrome is not always found. it starts with a sore throat due to pharyngitis. but it may not be that uncommon anymore [11]. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 159–162 161 the daily routine and especially in the emergent setting is limited [8]. tonsillitis. Tenderness develops along the lateral aspect of the sternocleidomastoid muscle representing thrombophlebitis of the internal jugular vein [6]. In addition. Written consent of the patient to publish his photographs. Contrast-enhanced CT scan is the diagnostic means of choice in the evaluation of the neck in Lemierre syndrome. which might rather have been a reactive inflammation due to facial vein thrombophlebitis rather than the bacterial entry point into the vessel. Lemierre syndrome was once called the “forgotten disease” because of its rarity. .8. especially affecting the lung [8]. The facial vein anatomically has contact with the submandibular gland.6]. Contrast-enhanced CT scan of the lung showing an infectious embolus (→). Our case is remarkable in as far as our patient had developed a thrombophlebitis limited to the facial vein—not the jugular vein. necrophorum infection. Kisser et al. necrophorum flourish. 3. for example.9]. its utility in Fig. Potential complications of facial vein thrombophlebitis are cavernous sinus thrombosis and septic embolism. Because isolation of F. taking into account that some Fusobacterium strains produce Fig. 4.U. Diagnosis is primarily clinical because culture of Fusobacterium species may take up to 6 to 8 days of incubation [1. Discussion Lemierre syndrome is a very rare disease and usually affects young healthy adults [1-5]. however. necrophorum is generally difficult. 5. Calculated intravenous antibiotic treatment with good anaerobic coverage needs to be started as soon as possible because postanginal sepsis is a potentially fatal condition with a mortality rate of about 6% and a rapid start of treatment is important for a good clinical outcome [1. Although magnetic resonance imaging scan might be equivalent to CT for diagnosis of Lemierre syndrome. followed by sepsis with high fever and shivering when anaerobic bacteria such as F. Lemierre syndrome should also be taken into account in case of negative blood culture results [4]. the original focus of infection remained unclear. and penetrate into the neighboring jugular vein [6-8]. Duong and Wenger [6] recommend metronidazole or aminopenicillin/sulbactam. in our case.

Prag J. Rev Infect Dis 1989. Summary • Lemierre syndrome is a rare disease in young healthy adults usually after a sore throat. Altozano JG. Incidence and clinical epidemiology of necrobacillosis. Lemierre's syndrome: a potentially fatal complication that may require vascular surgical intervention. [2] Moreno S. Lichtstein D.11:319-24. Because our patient is allergic to aminopenicillins. et al. Kisser et al.17:323-8. et al. After the start of antibiotic treatment. [11] Karkos PD. Asrani S. References [1] Leugers CM. Saxon P. On certain septicemias due to anaerobic organisms. [8] Weeks DF. et al.119:1552-9.91:901-9. metronidazole. • Severe facial infection with fever and general feeling of malaise after a sore throat should prompt the . Epidemiol Infect 1990. including Lemierre's syndrome. Laryngoscope 2009. Noakes DE. Pinilla B. but in cases of ongoing septic embolism. [9] Chirinos J. Eur J Clin Microbiol Infect Dis 1998. [10] Bartlett JG.21:589-93. Lancet 1936. Hild TG. J Vasc Surg 2005. Wallace LM. Garcia J.8:384-91.42:1023-5. clinician to consider Lemierre syndrome as a differential diagnosis to initiate timely. [7] Smith GR. Anaerobic bacterial infections of the lung. specific treatment. [3] Ramirez S. JABFM 1995. Wenger J. Increased diagnosis of Lemierre syndrome and other Fusobacterium necrophorum infections at a children's hospital. Lemierre's disease: postanginal bacteremia and pulmonary involvement caused by Fusobacterium necrophorum. In our case. [12] Charles K. Medicine 2002. 4. • This article describes an extremely rare case of Lemierre syndrome with facial vein thrombosis due to F. amoxicillin. [4] Hagelskjaer LH. In the current literature. Chest 1987. Experimental observations on the pathogenesis of necrobacillosis. and meropenem. Clover R. Surgical intervention is rarely needed. Katz DS. Lemierre's syndrome: a systematic review.17:561-5. The evolution of Lemierre syndrome: report of 2 cases and review of the literature. • It is caused by anaerobic fusobacteria and is characterized by jugular vein thrombosis and septic emboli throughout the body. Emerg Radiol 2010. necrophorum with prolonged facial swelling and septic emboli in the lung. Lemierre syndrome. piperacillin.10]. Pediatrics 2003.112:380-5. et al.11]. Lemierre syndrome: report of five new cases and literature review.104:73-8.162 U.8. microbiological testing revealed sensitivity of the bacteria to clindamycin.81:456-65. Pediatr Emerg Care 2005. Neschis DG. Flinn WE. Karkos CD. et al. there is no clear evidence of a benefit from anticoagulation therapy [6. in Denmark 1990–1995. [6] Duong M. Malxzynski J. Lemierre syndrome: postanginal sepsis. it may be necessary [12]. Rudolph C. [5] Lemierre A.1:701-3. et al. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 159–162 β-lactamase. we could observe a progression of clinical symptoms as well as a rise in the blood levels of inflammation markers before remission finally set in—a phenomenon already described by others and probably because of the exotoxins that are set free [6. he was treated with clindamycin and meropenem.