Journal of Otology & RhinologyISSN: 2324-8785

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Case Report, J Otol Rhinol Vol: 3 Issue: 2

Lemierre Syndrome in a Healthy Patient: A Late Complication of Tonsillectomy?

Annietta Gay and Lluís Nisa*
1Department of Otorhinolaryngology- Head and Neck Surgery, Geneva University Hospitals, Geneva, Switzerland
Corresponding author : Lluís Nisa
Department of Otorhinolaryngology- Head and Neck Surgery, Geneva University Hospitals, Rue Gabrielle-Gentil-Perret 4, 1121- Geneva- 14. Switzerland
Tel: +41-79-55-33922; Fax: +41-27-603-4449
E-mail: [email protected]
Received: December 15, 2013 Accepted: March 03, 2014 Published: March 10, 2014
Citation: Gay A, Nisa L (2014) Lemierre Syndrome in a Healthy Patient: A Late Complication of Tonsillectomy?. J Otol Rhinol 3:2. doi:10.4172/2324-8785.1000151

Abstract

Lemierre Syndrome in a Healthy Patient: A Late Complication of Tonsillectomy?

We present the case of an 18-year-old patient who developed bronchopneumonia shortly after tonsillectomy, for which she received a course of intervenous antibiotics. Almost two months after surgery she developed Lemierre syndrome (thrombophlebitis of the internal jugular vein and septic emboli of the lung). This complication of tonsillectomy seems to be rare, but should be considered in the differential diagnosis of the patient with respiratory infection in the post-tonsillectomy setting.

Keywords: Lemierre syndrome; Tonsillectomy; Postoperative complications

Keywords

Lemierre syndrome; Tonsillectomy; Postoperative complications

Introduction

Lemierre syndrome (LS) was described in 1936 by André Lemierre as a thrombophlebitis of the internal jugular vein with a potential for bacteriemia and septic emboli complicating tonsillitis or peritonsillar abscess. Usually due to Fusobacterium necrophorum, LS is considered rare nowadays in the antibiotic era, but its life-threatening course if not promptly and properly managed is still well acknowledged [1]. We report a case of LS in a young healthy patient occurring in the late postoperative period of a tonsillectomy and discuss the relevant literature.

Case Study

An 18-year-old female underwent elective tonsillectomy for recurring tonsillitis. The dissection was performed with cold-steel instruments and electrocoagulation. No particular difficulties were noted during the procedure and the patient was discharged after 24 hours. No antibiotic prophylaxis was given.
At postoperative day 3 the patient consulted her general practitioner for odynophagia, fever spikes, cough, and left lower chest pain. Physical examination showed an uncomplicated postoperative status and a clear pulmonary auscultation. Non-steroid antiinflammatory drugs were initially prescribed.
Without any improvement over the next 72 hours, a subsequent chest X-ray revealed pneumonia of the left inferior lobe. The patient was started on oral amoxicillin-clavulanate but did not improved clinically and remained febrile. She was admitted 48 hours later and started on intravenous amoxicilline-clavulanate. Puncture of a newlydeveloped pleural effusion yielded a Staphylococcus pettenkoferi. After infectious disease consultation, intravenous amoxicillinclavulanate was continued for 3 days and then orally for 7 days. Follow-up after one week showed an uneventful recovery.
At day 56 post-surgery the patient consulted the emergency room once more with left-sided neck pain and swelling, chest pain increasing during inspiration and coughing, and fever of up to 40°C. Two days before she was started on oral amoxicillin-clavulanate by her general practitioner. Doppler-ultrasonography showed a leftsided internal jugular vein thrombosis. Chest contrast-enhanced CTscan revealed three nodular lesions in the left inferior pulmonary lobe (Figure 1). The hemocultures did not show any growth.
Figure 1: Chest CT-scanner showing a nodular lesion of the lower left lobe (arrow), corresponding to a septic embolism.
Hospital treatment included intravenous amoxicillin and clindamyicin for a week, and anticoagulation with low molecular weight heparin (1 mg/kg twice a day). The treatment plan at discharge included oral clindamycin for 4 weeks and oral anticoagulation (acenocoumarol) for three months. Full clinical and ultrasonographic recovery persisted at 8 months follow-up.

Discussion

In the case we report the patient underwent tonsillectomy and shortly afterwards was diagnosed with a left-sided basal pneumonia. This pneumonia was firstly presumed to be a complication of intubation. Nearly 2 months later she developed a clinical picture featuring left-sided internal jugular vein thrombosis and three nodular lesions in the left inferior pulmonary lobe, a picture consistent with the diagnosis of Lemierre syndrome.
Reports of LS following tonsillectomy are seldom. The first case of LS following tonsillectomy was reported by Rubinstein et al. in 1974 [2]. More recently, Nix reported the case of a 16-year-old girl who developed LS at postoperative day 6, successfully managed with antibiotics but not anticoagulants [3]. Sagowski and Koch reported the case of a 62-year-old woman who developed LS at postoperative day 11 [4]. This patient underwent resection of the internal jugular vein, and was treated with antibiotics and anticoagulants.
Finally, Beldman et al. reported a 9-year-old boy that developed septic arthritis of the hip due to Fusobacterium necrophorum three days after tonsillectomy [5]. He underwent revision of the infected hip and was subsequently treated with antibiotics. The diagnosis in this child is not that of LS, but this case further illustrates the potential bacteriemic complications seen after tonsillectomy.
We were unable to identify further cases of LS developing in a post-tonsillectomy context through a Medline search.
Our case differs of these previous cases in that the diagnosis of LS was made almost two months after surgery, without any concomitant oropharyngeal infection. Cases of LS without oropharyngeal infection have not been reported to the best of our knowledge [1,3]. Nevertheless, our patient was treated with a course of intravenous antibiotics for pneumonia developing shortly after surgery. In retrospect, it could be speculated that this episode of pneumonia was the first manifestation of a LS, and that the antibiotic therapy partially decapitated subsequent evolution. Indeed, in 95% of the cases the presenting feature of LS is pulmonary: pleuritic pain, dyspnea or respiratory distress. The jugular thrombosis is initially silent in such cases [1-8].
Brocho-pulmonary infection and bacterial tracheitis have also been described following tonsillectomy, but such complications are most often attributed to the presence of an endotracheal tube during anesthesia [6]. Bacteriemia during tonsillectomy is often a transient phenomenon, without any clinical impact in otherwise healthy patients. Patients at risk include patients with congenital heart disease or with prosthetic material (valves, orthopedic), with the main complications being endocarditis and infection around the prosthetic material [7]. Experimental studies suggest that bacteriemia is caused by the germs present in the oropharyngeal flora, spread because of the surgical trauma [7].
In line with this, two pathophysiologic hypothesis for the development of LS after tonsillectomy can be suggested: 1) spread of tonsillar/peritonsillar infections to the parapharyngeal space with involvement of the jugular vein and development of a peri-infectious thrombophlebitis; and 2) colonization of the vascular endothelium after an episode of bacteriemia (the tonsillar fossae are drained by the lingual vein and the pharyngeal plexus, collaterals of the internal jugular vein) [1].
Fusobacterium necrophorum, an obligate anaerobic, non-motile, pleomorphic Gram-negative rod, is presumed to be part of the commensal flora of the oropharynx. It is by far the most commonly detected germ in cases of LS [1]. The recommended antibiotics against F. necrophorum are metronidazole or clindamycin [8].
In our case F. necrophorum was not identified. This could be due to the fact that our patient received a course of amoxicilline-clavulanate, to which this germ is at least partially sensitive [1,8], and in addition she was treated with clindamycin. The Staphylococcus pettenkoferi we identified in the pleural effusion liquid is rarely a cause of disease in otherwise healthy patients, but it is often a contaminating agent [9]. Although we cannot formally rule out the participation of this germ in the pathogenic process, it seems unlikely it played a role.
The prevention of infectious complications following tonsillectomy is an issue of controversy. In our institution patients operated for elective adeno-tonsillar surgery do not receive antibiotic prophylaxis unless they are considered at risk (mechanic cardiac valves, immunosuppression, orthopedic prosthesis, congenital heart disease). Indeed, current evidence does not allow systematic recommendation of antibiotic prophylaxis for tonsillectomy [10].
The management of LS is also controversial and somehow poorly characterized, doubtless because it remains a rare condition. While large-spectrum antibiotics covering F. necrophorum are universally advocated in the management of LS, the indications for anticoagulation or resection of the thrombotic vein are not well defined [1].

Conclusion

Even though severe infectious complications are rarely seen following tonsillectomy, these must be acknowledged and early identified to avoid their potentially-fatal complications. Lemierre syndrome is scarcely reported following tonsillectomy, but should be considered in the differential diagnosis of the patient with pulmonary manifestations in the post-tonsillectomy period.

Acknowledgments

The authors wish to acknowledge the skillful assistance of Mrs Irina Ciorba- Nisa, MS, in the preparation of this manuscript.

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