Case Report

Lemierre Syndrome Caused by Streptococcus constellatus Mono-Infection: A Case Report

Da-Hee Park1https://orcid.org/0000-0001-7466-2793, Jin-Choon Lee1,2https://orcid.org/0000-0002-4056-8763, Hwan-Jung Roh1,2,*https://orcid.org/0000-0002-3055-8713
Author Information & Copyright
1Department of Otorhinolaryngology-Head and Neck Surgery, Pusan National University Yangsan Hospital, Yangsan, Korea
2Department of Otorhinolaryngology-Head and Neck Surgery, School of Medicine, Pusan National University, Busan, Korea
*Corresponding author: Hwan-Jung Roh, Department of Otorhinolaryngology-Head and Neck Surgery, Pusan National University Yangsan Hospital, Yangsan 50612, Korea, Tel: +82-55-360-1440, Fax: +82-55-360-2930, E-mail: rohhj@pusan.ac.kr

© Copyright 2023 xxxxxxxxxxxxxxx. This is an Open-Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Received: Oct 25, 2023; Revised: Nov 30, 2023; Accepted: Dec 12, 2023

Published Online: Dec 31, 2023

ABSTRACT

The onset of Lemierre syndrome is marked by oral or oropharyngeal infection, especially a parapharyngeal abscess. Patients having Lemierre syndrome often do not present with severe throat symptoms, making clinical diagnosis difficult. The prognosis is poor if the patient is not promptly diagnosed and actively treated. Herein, we describe a patient who developed Lemierre syndrome following a peritonsillar abscess. Blood tests revealed septicemia and computed tomography showed an internal jugular vein thrombosis. The patient was immediately underwent incision and drainage and Streptococcus constellatus was cultured. Antibiotics were administered for a month postoperatively and the patient recovered successfully. This case represents successful outcome of Lemierre syndrome with Streptococcus constellatus mono-infection, which is a relatively rare course of Lemierre syndrome.

Keywords: Lemierre syndrome; Thrombophlebitis; Septicemia; Deep neck infection; Case report

Introduction

In 1936, Andre Lemierre reported throat infections, especially anaerobic septicemia after a peritonsillar abscess, and abscess formation in various areas, such as the lungs and joints.1) These findings are referred to as the Lemierre syndrome, which is characterized by thrombophlebitis of the internal jugular vein (IJV), septicemia, and septic emboli that spread to various organs, such as the lungs.1) Infectious bacteria are mainly anaerobic, and Fusobacterium necrophorum is the most common infectious bacterium causing Lemierre syndrome.

Despite the low incidence, Lemierre syndrome is a severe disease that progresses quickly, invades various organs, and causes septicemia. In the 2000s, the mortality rate is reported about 5%, which is significantly lower than the mortality rate of 90% initially reported in 1936.1,2) This drop in mortality is attributed to the development of antibiotics. Although the prevalence of Lemierre syndrome has been continuously declining since the advent of antibiotics, a recent spike has been observed. This increase is likely related to the increase in antibiotic-resistant bacteria.3) Therefore, understanding Lemierre syndrome is essential for the successful treatment of this disease despite the low number of cases encountered in clinical practice.

In this paper, we report the successful surgical drainage and postoperative treatment of a patient with Lemierre syndrome due to Streptococcus constellatus.

Case Report

A 51-year-old female patient complained of fever and left neck pain lasting for 1 week. She was admitted to another hospital and prescribed antibiotic treatment, but her symptoms did not improve and progressed to thrombocytopenia and septicemia. Therefore, she was transferred to the emergency room for further evaluation and treatment.

Swelling on the left side of the neck was observed on physical examination. Left peritonsillar swelling was observed on laryngoscopy, and her airway was patent. Her vital signs were as follows: body temperature, 37.7℃; systolic blood pressure, 70 mmHg; and diastolic blood pressure, 40 mmHg. Blood tests revealed thrombocytopenia (platelet count of 12×103/µL [reference: 140×103–400×103/µL]), leukocytosis (white blood cell count of 25.44×103/µL [reference: 4.0×103–11.0×103/µL] and neutrophil segment of 92% [reference: 40%–73%]), and a high sensitivity C-reactive protein level of 20.31 mg/dL (reference: 0–0.5 mg/dL). The blood culture was positive for Clostridium clostridioforme approximately 2 days after admission. Therefore, septicemia due to an uncontrolled pharyngolaryngeal infection was suspected.

Contrast-enhanced computed tomography (CT) showed a peritonsillar abscess and thrombophlebitis of the left IJV (Fig. 1). As there was uncontrolled septicemia and thrombophlebitis of the IJV, we decided to perform a transcervical drainage rather than an intraoral drainage to obtain a wider surgical field and to remove the thrombosis inside IJV. Emergency abscess drainage was performed under general anesthesia. The abscess was observed along the left IJV from the mandible to the clavicle. The tearing of left facial vein was occurred during thrombectomy and facial vein was sutured with a 6-0 prolene.

jcohns-34-4-240-g1
Fig. 1. CT neck contrast enhanced. A, C: A left peritonsillar abscess with gas formation was confirmed on a contrast-enhanced computed tomography scan confirmed (single arrows). B, D: Thrombophlebitis with an air bubble that occurred along the internal jugular vein was identified (double arrows). Thrombosis exists between the tunica intima and the tunica externa of the internal jugular vein.
Download Original Figure

Strepcococcus constellatus was cultured and it was susceptible to all antibiotics. Piperacillin/tazobactam and levofloxacin were used as antibiotics for 22 days after surgery.

After the surgery, the patient was admitted to the intensive care unit for treatment. A postoperative day 3 contrast-enhanced chest CT revealed diffuse airspace consolidations in the bilateral lower lobes and multiple patchy areas of glassy opacities of the lungs suggestive of septic pneumonia and bilateral pleural effusions. The pneumonia improved with continuous piperacillin/tazobactam and levofloxacin therapy. On postoperative day 8, she was transferred to a general ward and safely discharged on day 25.

Discussion

The onset of Lemierre syndrome is marked by oropharyngeal infection with symptoms such as a sore throat, fever, and cervical tenderness and swelling. Parapharyngeal abscess formation and peripheral suppurative thrombophlebitis in the IJV occur, causing complications such as septicemia, carotid artery rupture, and pulmonary embolism; these conditions affect other organs, such as the joints, kidneys and lungs.4)

The prevalence of Lemierre syndrome has gradually decreased due to the development of antibiotics, with an incidence between 0.6 and 2.3 per million people.4) In addition, the overall mortality rate is reported to be 4%–18%, 70% of patients are young adults between the ages of 16 and 25 years, and twice as many cases involve males compared with females.2,4) According to Karkos et al., approximately 200 papers on Lemierre syndrome were published from 1980 to 2010, and the number tended to increase with time, most likely due to increase in the number of antibiotic-resistant bacteria and newly developed diagnostic technology.3,5)

In general, Fusobacterium necrophorum is the most common bacteria causing Lemierre syndrome, accounting for about 62.0%–81.7% of cases. Streptococcus spp. and Staphylococcus spp. also cause Lemirre syndrome.4,6) In the present case, Streptococcus constellatus was cultured. Streptococcus constellatus was reported to cause neck abscesses and IJV thrombosis in 25% of Lemierre cases.7) It is reported that a mixed infection of Fusobacterium and Streptococcus is more dangerous than either alone.8) Therefore, if Streptococcus is identified, it is necessary to confirm that it is not co-infected with Fusobacterium. In this case, Streptococcus was identified as an isolated infection.

Diagnostic criteria for Lemierre syndrome have not yet been clearly established, but in 1989, Sinave et al. proposed diagnostic criteria based on characteristic symptoms: primary infection of the oropharynx, sepsis caused by one or more bacteria streaked on blood culture, IJV thrombosis, and involvement of other organ.9)

Contrast-enhanced CT is the most important tool for diagnosis, where abscess formation in the parapharyngeal area, a filling defect in the IJV, and septic pulmonary emboli can be observed. In addition to the IJV, there are cases where adjacent blood vessels, such as the superior ophthalmic vein, facial vein, and sigmoid sinus, are affected.10) The extent of the disease and the invading blood vessels need to be evaluated before surgical treatment.

Treatment of Lemierre syndrome is based on the use of antibiotics, and surgical treatment is performed when the lesion is localized and the patient's condition is operable. In general, penicillin, metronidazole, and second- and third-generation cephalosporins are used for 3–6 weeks or longer.11) However, as the number of antibiotic-resistant bacteria are increasing and bacteria other than Fusobacterium necrophorum can be an infection source, it is important to identify the strain of bacteria and determine the appropriate antibiotics.

As the main symptoms are of septic thrombophlebitis and septic emboli of the IJV, anticoagulants are sometimes used for treatment.12) Although many studies have been conducted on the use of anticoagulants, it is still unclear whether they are effective. After starting treatment, patients require an average hospitalization of about one month, and 58% of patients receive treatment in the intensive care unit.2)

In the case presented herein, the patient underwent immediate surgery with broad spectrum antibiotic treatment. Compared to 77.6% of immediate surgical drainage in patients with common deep neck infections, the rate of surgical drainage is relatively low for patients with Lemierre syndrome.8,13) Invasion and distant infection of the IJV and adjacent blood vessels and septicemia may limit the use of general anesthesia. Surgery helps with rapid recovery, but it is important to accurately grasp the extent of the lesion before the procedure.

Conclusion

Lemierre syndrome is rare but it could be fatal which requires prompt diagnosis and treatment. Contrast-enhanced CT is important to decide the extent of the lesion and culture-based antibiotics should be used immediately.

It seems that Streptococcus constellatus mono-infection is related to better prognosis combined with early surgical intervention including drainage and thrombectomy of IJV.

Acknowledgements

Not applicable.

Funding Information

This work was supported by a 2-Year Research Grant of Pusan National University.

Conflicts of Interest

No potential conflict of interest relevant to this article was reported.

Author Contribution

Conceptualization: Roh HJ.

Data curation: Lee JC.

Formal analysis: Park DH.

Methodology: Roh HJ.

Validation: Lee JC.

Investigation: Lee JC.

Writing - original draft: Park DH.

Writing - review & editing: Park DH, Lee JC, Roh HJ.

Ethics Approval

Research was conducted according to all ethical standards. This study was approved by the Institutional Review Board (IRB) of the Pusan Nat’l University Yangsan Hospital, Korea (Approval No. 05-2021-024).

References

1.

Lemierre A. On certain septicaemias due to anaerobic organisms. Lancet 1936;227(5874):701-3.

2.

Karkos PD, Asrani S, Karkos CD, Leong SC, Theochari EG, Alexopoulou TD, et al. Lemierre’s syndrome: a systematic review. Laryngoscope 2009;119(8):1552-9.

3.

Harper LK, Pflug K, Raggio B, April D, Milburn JM. Clinical images: Lemierre syndrome: the forgotten disease? Ochsner J 2016;16(1):7-9.

4.

Chirinos JA, Lichtstein DM, Garcia J, Tamariz LJ. The evolution of Lemierre syndrome: report of 2 cases and review of the literature. Medicine 2002;81(6):458-65.

5.

Tsai YJ, Lin YC, Harnnd DJ, Chiang RPY, Wu HM. A Lemierre syndrome variant caused by Klebsiella pneumoniae. J Formos Med Assoc 2012;111(7):403-5.

6.

Valerio L, Zane F, Sacco C, Granziera S, Nicoletti T, Russo M, et al. Patients with Lemierre syndrome have a high risk of new thromboembolic complications, clinical sequelae and death: an analysis of 712 cases. J Intern Med 2020;289(3):325-39.

7.

Hahn J, Nordmann-Kleiner M, Hoffmann TK, Greve J. Thrombosis of the internal jugular vein in the ENT-department — prevalence, causes and therapy: a retrospective analysis. Auris Nasus Larynx 2019;46(4):624-9.

8.

Kuriyama T, Nakagawa K, Kawashiri S, Yamamoto E, Nakamura S, Karasawa T. The virulence of mixed infection with Streptococcus constellatus and Fusobacterium nucleatum in a murine orofacial infection model. Microbes Infect 2000;2(12):1425-30.

9.

Sinave CP, Hardy GJ, Fardy PW. The Lemierre syndrome: suppurative thrombophlebitis of the internal jugular vein secondary to oropharyngeal infection. Medicine 1989;68(2):85-94.

10.

Akiyama K, Karaki M, Samukawa Y, Mori N. Blindness caused by septic superior ophthalmic vein thrombosis in a Lemierre syndrome variant. Auris Nasus Larynx 2013;40(5):493-6.

11.

Kuppalli K, Livorsi D, Talati NJ, Osborn M. Lemierre’s syndrome due to Fusobacterium necrophorum. Lancet Infect Dis 2012;12(10):808-15.

12.

Phua CK, Chadachan VM, Acharya R. Lemierre syndrome—should we anticoagulate? A case report and review of the literature. Int J Angiol 2013;22(02):137-42.

13.

Velhonoja J, Lääveri M, Soukka T, Irjala H, Kinnunen I. Deep neck space infections: an upward trend and changing characteristics. Eur Arch Otorhinolaryngol 2020;277(3):863-72.

34권 4호 Quiz 정답 및 해설, 당첨자 발표


정답 ②

- 소음환경에서의 청취 편의성과 음성 명료성을 위하여 
디지털 신호 처리 기술이 사용되고 있는데 
그중 대표적인 것이 디지털 소음 감소(digital noise reduction, DNR) 기능과  
방향성 기술(directionality) 사용이다.

- 미국의 Food and Drug Administration(FDA)의 정의에 따르면 PSAP는 
단순히 소리를 증폭하는데 사용되며, 정상 청력을 가진 모든 연령의 
일반인에게 특정상황에서 소리를 증폭하는데에 사용되는 일반 전자제품이다. 
하지만 최근의 제품들은 경도와 중등도 난청 환자에서 보청기와 유사한 효과를 
보인다는 연구 결과를 보이기도 하였다. 아직 고도난청 환자에게 PSAP 를 
적용하는 것에 대한 효과는 증명되어 있지 않다.

- 최신 보청기에는 심박동 센서와 움직임 센서를 탑재 하여 걸음 수, 심박동변화, 
및 착용시간을 추적관리 할 수 있도록 하였고, 다른 모델에서는 움직임 센서를
기반으로 하여  낙상을 감지하여 사용자나 사용자의 가족에게 통보해 줄 수 있는 
기능을 탑재 하였다.

- 저전력 블루투스 기술을 사용할 경우 더 낮은 전력소모량으로 
더 나은 음질을 확보할 수 있다.
 

당첨자(5명)

서*현    010-****-**77
송*화    010-****-**24
이*미    010-****-**78
이*희    010-****-**44
최*홍    010-****-**90

 

응모해 주신 모든분들께 감사드립니다.

경품은 당첨자분들께 2.21(수)까지 개별 발송해 드리겠습니다.

 

임상이비인후과 편집위원회 


I don't want to open this window for a day.