Lemierre’s syndrome as a rare cause of septic pulmonary embolism (case report)

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Abstract

Objectives. The purpose of this work is to provide a clinical case study of Lemierre’s syndrome (LS) in order to raise awareness amongst doctors of various medical specialties regarding this rare cause of septic pulmonary embolism (SPE).

Materials and methods. The patient in clinical case is a 25-year-old female who was hospitalized due to SPE, which had been complicated by respiratory distress, pneumothorax and septic shock. The initial source of embolus lung damage was the septic thrombosis of both the internal and external jugular veins. This thrombosis occurred against the backdrop of a carbuncle of the lower lip, an inflammatory infiltrate at the bottom of the mouth, an abscess in the soft tissue of the chin and neck cellulitis, which correspond to the diagnostic criteria for Lemierre Syndrome.

Results. The literature data of the epidemiology, etiology, pathogenic mechanisms of the venous thrombosis, embolic and septic complications among patients with LS are presented. The article discusses the clinical features and course of the disease, as well as key aspects of diagnosis, including current diagnostic criteria, with regard to the typical and atypical variants of LS. The principles of antibiotic therapy and the possibility of anticoagulation therapy in cases of septic pulmonary emboli associated with head and neck vein thrombosis are also considered.

Conclusion. LS is a rare, but clinically significant condition characterized by the development of septic thrombosis in the internal jugular vein in response to acute suppurative inflammation of the head and neck. In typical cases, the causative organism is Fusobacterium necrophorum, or other anaerobic bacteria. This pathological condition is often accompanied by multiple other septic complications, with the most significant being the development of septic pulmonary embolism. Clinical observation indicates the necessity for including LS in the differential diagnosis among causes of the septic pulmonary embolism, and for adopting a multidisciplinary approach in the treatment of these patients in a specialized multifield hospital. Identification and diagnosis of Lemierre Syndrome can be challenging due to its low incidence and variable clinical presentation. Although the condition is not widely recognized among physicians, an understanding of the basic principles of its diagnosis and management can significantly improve treatment outcomes and patients prognosis.

About the authors

N. S. Chipigina

N.I. Pirogov National Research Medical University, Ministry of Health of Russia

Author for correspondence.
Email: chipigina-natalia56@yandex.ru
ORCID iD: 0000-0002-2083-0437

Natalia Semyonovna Chipigina 

1 Ostrovityanova St., Moscow 117997

 

Russian Federation

N. Yu. Karpova

N.I. Pirogov National Research Medical University, Ministry of Health of Russia

ORCID iD: 0000-0002-7546-4841

1 Ostrovityanova St., Moscow 117997

Russian Federation

A. S. Vinokurov

N.I. Pirogov National Research Medical University, Ministry of Health of Russia;
V.P. Demikhov City Hospital, Moscow Healthcare Department;
Moscow Multidisciplinary Clinical Center “Kommunarka”, Moscow Healthcare Department

ORCID iD: 0000-0002-0745-3438

1 Ostrovityanova St., Moscow 117997;
4 Shkuleva St., Moscow 109263;
8 Sosenskiy stan St., Kommunarka, 108814 Moscow

Russian Federation

D. P. Kotova

N.I. Pirogov National Research Medical University, Ministry of Health of Russia;
N.I. Pirogov City Clinical Hospital No. 1, Moscow Healthcare Department

ORCID iD: 0000-0003-1071-0877

1 Ostrovityanova St., Moscow 117997;
8 Leninskiy Avenue, Moscow 119049

Russian Federation

A. A. Gasparyan

N.I. Pirogov National Research Medical University, Ministry of Health of Russia

ORCID iD: 0000-0003-1699-7717

1 Ostrovityanova St., Moscow 117997

Russian Federation

P. A. Kashkovskaya

N.I. Pirogov National Research Medical University, Ministry of Health of Russia

ORCID iD: 0009-0001-0856-9504

1 Ostrovityanova St., Moscow 117997

Russian Federation

A. G. Makaev

N.I. Pirogov National Research Medical University, Ministry of Health of Russia

ORCID iD: 0000-0003-2628-2440

1 Ostrovityanova St., Moscow 117997

Russian Federation

M. S. Sapko

N.I. Pirogov National Research Medical University, Ministry of Health of Russia

ORCID iD: 0009-0009-8151-8023

1 Ostrovityanova St., Moscow 117997

Russian Federation

References

  1. Lemierre A. On certain septicaemia due to anaerobic organisms. Lancet 1936;227(5874):701–3. doi: 10.1016/S0140-6736(00)57035-4
  2. Apostilidi K.G., Savchuk O.V., Epifanov S.A., Isaeva M.L. The forgotten syndrome of Lemierre. Vestnik N.I. Pirogova Nacional’nogo mediko-hirurgicheskogo centra = Bulletin of N.I. Pirogov National Medical and Surgical Center 2019; 14(1):153–7. (In Russ.). doi: 10.25881/BPNMSC.2019.96.35.029
  3. Tiwari A. Lemierre’s Syndrome in the 21st Century: A literature review. Cureus 2023;15(8):e43685. doi: 10.7759/cureus.43685
  4. Courmont P., Cade A. Sur une septico-pyohemie de I’homme stimulant la peste et causee par un streptobacille anaerobie. Arch Med Exp Anat Pathol 1900;12(4):393–418.
  5. Mosher H.P. Deep cervical abscess and thrombosis of the internal jugular vein. Laryngoscope 1920;30(6):365–75. doi: 10.1288/00005537-192006000-00005
  6. Shannon G.W., Ellis C.V., Stepp W.P. Oropharyngeal bacteroides melaninogenicus infection with septicemia: Lemierre’s syndrome. J Fam Pract 1983;16(1):159–60, 163, 166.
  7. Valerio L., Corsi G., Sebastian T., Barco S. Lemierre syndrome: current evidence and rationale of the bacteria-associated thrombosis, thrombophlebitis and Lemierre syndrome (BATTLE) registry. Thromb Res 2020;196:494–9. doi: 10.1016/j.thromres.2020.10.002
  8. Riordan T. Human infection with Fusobacterium necrophorum (Necrobacillosis), with a focus on Lemierre’s syndrome. Clin Microbiol Rev 2007;20(4):622–59. doi: 10.1128/CMR.00011-07.
  9. Sinave C.P., Hardy G.J., Fardy P.W. The Lemierre syndrome – suppurative thrombophlebitis of internal jugular vein secondary to oropharyngeal infection. Medicine (Baltimore) 1989;68(2):85–94.
  10. Van Hoecke F., Lamont B., Van Leemput A., Vervaeke S. A Lemierre-like syndrome caused by Staphylococcus aureus: an emerging disease. Infect Dis (Lond) 2020;52(3):143–51. doi: 10.1080/23744235.2019.1691255
  11. Lee W.S., Jean S.S., Chen F.L. et al. Lemierre’s syndrome: a forgotten and re-emerging infection. J Microbiol Immunol Infect 2020;53(4):513–7. doi: 10.1016/j.jmii.2020.03.027
  12. Righini C.A., Karkas A., Tourniaire R. et al. Lemierre syndrome: study of 11 cases and literature review. Head Neck 2014;36(7):1044–51. doi: 10.1002/hed.23410
  13. Ramirez S., Hild T.G., Rudolph C.N. et al. Increased diagnosis of Lemierre syndrome and other Fusobacterium necrophorum infections at a Children’s Hospital. Pediatrics 2003;112(5):e380. doi: 10.1542/peds.112.5.e380.
  14. Pokharel A., Basnet P., Sharma B., Thapa K. Lemierre Syndrome. A Forgotten Disease. Case report and review of literature. Int Med Case Rep J 2019;12:367–71. doi: 10.2147/IMCRJ.S218895
  15. Nur Iyow S., Uzel M., Ibrahim I.G. et al. Lemierre Syndrome: incidental finding of forgotten fatal disease as a complication of Ludwig’s angina. Open Access Emerg Med 2023;15:259–63. doi: 10.2147/OAEM.S408470
  16. Nygren D., Holm K. Invasive infections with Fusobacterium necrophorum including Lemierre’s syndrome: an 8-year Swedish Nationwide Retrospective Study. Clin Microbiol Infect 2020;26(8):1089.e7–12. doi: 10.1016/j.cmi.2019.12.002
  17. Hagelskjaer Kristensen L., Prag J. Lemierre’s syndrome and other disseminated Fusobacterium necrophorum infections in Denmark: a prospective epidemiological and clinical survey. Eur J Clin Microbiol Infect Dis 2008;27(9):779–89. doi: 10.1007/s10096-008-0496-4
  18. Hagelskjaer L.H., Prag J., Malczynski J., Kristensen J.H. Incidence and clinical epidemiology of necrobacillosis, including Lemierre’s syndrome, in Denmark 1990–1995. Eur J Clin Microbiol Infect Dis 1998;17(8):561–5. doi: 10.1007/BF01708619
  19. Valerio L., Zane F., Sacco C. 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 2021;289(3):325–39. doi: 10.1111/joim.13114
  20. Root R.W., Barrett T.W., Abramo T.J. A 10-month-old with Lemierre syndrome complicated by purulent pericarditis. Am J Emerg Med 2013;31(1):274.e5–7. doi: 10.1016/j.ajem.2012.05.019
  21. Suzuki T., Kojima M., Nakamoto R. et al. A case of blindness caused by Lemierre’s syndrome. Clin Case Rep 2023;11(12):e8327. doi: 10.1002/ccr3.8327
  22. Pleming W., Barco S., Voci D. et al. Cardiac and cerebral arterial complications of Lemierre syndrome: results from a Systematic Review and individual patient data meta-analysis. Hamostaseologie 2022;42(4):261–7. doi: 10.1055/a-1694-8723
  23. Kuppalli K., Livorsi D., Talati N.J., Osborn M. Lemierre’s syndrome due to Fusobacterium necrophorum. Lancet Infect Dis 2012;12(10):808–15. DOI: 10.1-16/S1473-3099(12)70089-0
  24. Alifano M., Venissac N., Guillot F., Mouroux J. Lemierre’s syndrome with bilateral empyema thoracis. Ann Thorac Surg 2000;69(3):930–1. doi: 10.1016/s0003-4975(99)01404-6
  25. De Smet K., Claus P.E., Alliet G. et al. Lemierre’s syndrome: a case study with a short review of literature. Acta Clin Belg 2019;74(3):206–10. doi: 10.1080/17843286.2018.1474614
  26. Lanfear A.T., Hamandi M., Fan J. et. al. Lemierre’s syndrome treated operatively. Proc (Bayl Univ Med Cent) 2020;33(4):671–3. doi: 10.1080/08998280.2020.1772011
  27. Prasad R.M., Raziq F., Kemnic T., Abubaker A. Implantable Port Developing Septic Pulmonary Emboli and Secondary Spontaneous Pneumothorax. Am J Med Case Rep 2021;9(1):35–9. doi: 10.12691/ajmcr-9-1-10
  28. Ye R., Zhao L., Wang C. et al. Clinical characteristics of septic pulmonary embolism in adults: a systematic review. Respir Med 2014;108(1):1–8. doi: 10.1016/j.rmed.2013.10.012
  29. Сhipigina N.S., Karpova N.Yu., Vinokurov A.S. et al. Septic embologenic pneumonia – clinical and diagnostical features (review and own observations). Arkhiv vnutrennej meditciny = The Russian Archives of Internal Medicine 2023;13(4):272–81. (In Russ.). doi: 10.20514/2226-6704-2023-13-4-272-281
  30. Jing J., Liang Q.L, Liu L.H. et al. Septic pulmonary embolism in China: clinical features and analysis of prognostic factors for mortality in 98 cases BMC Infect Dis 2019;19(1):1082. doi: 10.1186/s12879-019-4672-1
  31. Goswami U., Brenes J.A., Punjabi G.V. et al. Associations and outcomes of septic pulmonary embolism. Open Respir Med J 2014;8:28–33. doi: 10.2174/1874306401408010028
  32. Brenes J.A., Goswami U., Williams D.N. The association of septic thrombophlebitis withseptic pulmonary embolism in adults. Open Respir Med J 2012;6:14–9. doi: 10.2174/1874306401206010014
  33. Goldenberg N.A., Knapp-Clevenger R., Hays T., Manco-Johnson M.J. Lemierre’s and Lemierre’s-like syndromes in children: survival and thromboembolic outcomes. Pediatrics 2005;116(4):e543–8. doi: 10.1542/peds.2005-0433
  34. Sacco C., Zane F., Granziera S. et al. Lemierre Study Group. Lemierre syndrome: Clinical update and protocol for a systematic review and individual patient data meta-analysis. Hamostaseologie 2019;39(1):76–86. doi: 10.1055/s-0038-1654720
  35. Venditto L., Ferrante G., Caccin A. et al. Lung abscess as a complication of Lemierre syndrome in adolescents: a single center case reports and review of the literature. Ital J Pediatr 2023;49(1):96. doi: 10.1186/s13052-023-01499-4
  36. Elhakeem I.A., Al Shokri S.D., Elzouki A.Y., Danjuma M.I. An unusual case of modified Lemierre’s syndrome caused by Staphylococcus aureus cellulitis. Am J Case Rep 2020;21:e916575. doi: 10.12659/AJCR.916575
  37. Zamora Gonzalez R.A., Sarria J.C., Christians N.A., Baliss M. Lemierre’s syndrome due to methicillin-resistant Staphylococcus aureus. J Investig Med High Impact Case Rep 2019;7:2324709619890967. doi: 10.1177/2324709619890967
  38. Kherabi Y., Chevrel G., Roux D., Federici L. Syndrome de Lemierre inversé: à propos d’un cas et revue de la littérature [Gynecological Lemierre’s syndrome: A case report and literature review]. Rev Med Interne 2020;41(7):493–5. (In French.). doi: 10.1016/j.revmed.2020.02.012
  39. Ridgway J.M., Parikh D.A., Wright R. et al. Lemierre syndrome: a pediatric case series and review of literature. Am J Otolaryngol 2010;31(1):38–45. doi: 10.1016/j.amjoto.2008.09.006
  40. Severiche-Bueno D.F., Insignares-Niño D.A., Severiche-Bueno D.F. et al. Lemierre’s syndrome by Bacillus circulans, Fusobacterium nucleatum and Staphylococcus aureus with involvement of the internal and external jugular vein. Germs 2021;11(2):314–18. doi: 10.18683/germs.2021.1267
  41. Chirinos J.A., Lichtstein D.M., Garcia J., Tamariz L.J. The evolution of Lemierre’s syndrome: report of 2 cases and review of the literature. Medicine (Baltimore) 2002;81(6):458–65. doi: 10.1097/00005792-200211000-00006
  42. Johannesen K.M., Bodtger U. Lemierre’s syndrome: current perspectives on diagnosis and management. Infect Drug Resist 2016;9:221–7. doi: 10.2147/IDR.S95050
  43. Chuncharunee A., Khawcharoenporn T. Lemierre’s syndrome caused by Klebsiella pneumoniae in a diabetic patient: A case report and review of the literature. Hawaii J Med Public Health 2015;74(8):260–6.
  44. Chen T.A., Chuang Y.T., Lin H.Y., Chen C.H. Lemierre’s syndrome caused by Klebsiella pneumoniae: a case report and literature review. Cureus 2023;15(8):e44434. doi: 10.7759/cureus.44434
  45. Holm K., Frick I.M., Björck L., Rasmussen M. Activation of the contact system at the surface of Fusobacterium necrophorum represents a possible virulence mechanism in Lemièrre’s syndrome. Infect Immun 2011;79(8):3284–90. doi: 10.1128/IAI.05264-11
  46. Nygren D., Elf J., Torisson G., Holm K. Jugular vein thrombosis and anticoagulation therapy in Lemierre’s syndrome – A post hoc observational and population-based study of 82 patients. Open Forum Infect Dis 2020;8(1):ofaa585. doi: 10.1093/ofid/ofaa585
  47. Nygren D., Torisson G., Happonen L. et al. Proteomic characterization of plasma in Lemierre’s syndrome. Thromb Haemost 2024;124(5):432–40. doi: 10.1055/a-2195-3927
  48. Holm K., Svensson P.J., Rasmussen M. Invasive Fusobacterium necrophorum infections and Lemièrre’s syndrome: the role of thrombophilia and EBV. Eur J Clin Microbiol Infect Dis 2015;34(11):2199–207. doi: 10.1007/s10096-015-2469-8
  49. Malmberg S., Petrén S., Gunnarsson R. et al. Acute sore throat and Fusobacterium necrophorum in primary healthcare: a systematic review and meta-analysis. BMJ Open 2021;11(6):e042816. doi: 10.1136/bmjopen-2020-042816
  50. Gore M.R. Lemierre Syndrome: A meta-analysis. Int Arch Otorhinolaryngol 2020;24(3):e379–85. doi: 10.1055/s-0039-3402433
  51. Bahall M., Giddings S., Bahall K. Lemierre’s syndrome: forgotten, but not absent. BMJ Case Rep 2017;2017:bcr2017221203. doi: 10.1136/bcr-2017-221203
  52. Bourgeaud J., Delabays B., Van den Bogaart L., Ratano D. Complex Lemierre syndrome with multisystemic abscesses. BMJ Case Rep 2023;16(4):e254638. doi: 10.1136/bcr-2023-254638
  53. Thurnheer T., Bensland S., Eick S. et al. Antibiotic resistance among Fusobacterium, Capnocytophaga, and Leptotrichia species of the oral cavity. Oral Health Prev Dent 2023;21(1):93–102. doi: 10.3290/j.ohpd.b4009553
  54. Cupit-Link M.C., Nageswara Rao A., Warad D.M., Rodriguez V. Lemierre Syndrome: a retrospective study of the role of anticoagulation and thrombosis outcomes. Acta Haematol 2017;137(2):59–65. doi: 10.1159/000452855
  55. Stevens S.M., Woller S.C., Kreuziger L.B. et al. Antithrombotic therapy for VTE disease: second update of the CHEST guideline and expert panel report. Chest 2021;160(6):e545–608. doi: 10.1016/j.chest.2021.07.055.

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