Lemierre's syndrome due to Klebsiella pneumoniae in a 63-year-old man with diabetes: a case report

Abstract

IntroductionLemierre's syndrome was originally documented to be caused by Fusobacterium necrophorum. It is a very rare condition with a prevalence of one to 14.4 instances per million. Its presentation is varied, not only in composition but also in the infecting organism. Treatment with anticoagulants has been controversial and applied only on a case-by-case basis.Case presentationA 63-year-old Saudi man who had had uncontrolled diabetes mellitus for 47 years presented to our facility with a five-day history of swelling on the right side of his neck and fever. The swelling progressively increased in size and was associated with pain, dysphagia, odynophagia, change of voice ('hot potato voice'), and reduced appetite. Abscess content culture and sensitivity testing revealed Klebsiella pneumoniae. However, blood culture results were repeatedly negative. The abscess was incised and drained without any complication. Our patient was treated with clindamycin and cefuroxime. Warfarin was also administered concurrently for six weeks, for an isolated internal jugular vein thrombosis (IJV), with complete resolution of the thrombus. Normoglycemia was achieved and our patient was discharged after complete wound healing and the return of his biochemical parameters to normal.ConclusionsOnly two cases of Lemierre's syndrome in patients with diabetes due to K. pneumoniae have been reported previously. A review of the literature suggested that an association exists between deep neck infections due to K. pneumoniae and diabetes mellitus. The reasons for this association are still not clear. This poses a question as to whether diabetes mellitus specifically predisposes these patients to infection with this organism. It is suggested that clinicians should consider infectious agents other than F. necrophorum in the causation of Lemierre's syndrome, especially in patients with diabetes.

Lemierre Syndrome—Should We Anticoagulate? A Case Report and Review of the Literature

“…Although classically F. necrophorum has been described in young patients with Lemierre syndrome, there have been increasing reports of K. pneumoniae being isolated in older patients with poorly controlled diabetic mellitus. [4][5][6] This is an interesting observation although the reason for this is not clear. Locally, it has been reported that K. pneumoniae accounted for 27.1% of pathogens cultured from deep neck abscesses, of which half had diabetes mellitus.…”

“…It has been argued that thrombosis associated with Lemierre syndrome will spontaneously resolve, but it is also unclear if anticoagulation will expedite the resolution of thrombosis. 9 Many have reported successful treatment in both patients with 4,6,[12][13][14][15] or without anticoagulation 2,[16][17][18][19] in conjunction with antimicrobial therapy (►Table 1). In a small retrospective study of 11 patients with internal jugular venous thrombosis associated with deep neck infection in intravenous drug abusers, there were no serious adverse consequences in those patients who did not receive anticoagulation.…”

“…However, there are increasing reports of Lemierre syndrome with Klebsiella pneumoniae in the context of poorly controlled diabetes mellitus. [4][5][6] Treatment typically involves prompt antimicrobial therapy and surgical drainage of accessible infected collections, but the role of anticoagulation is controversial due to the lack of controlled studies.…”

Lemierre Syndrome: A Retrospective Study of the Role of Anticoagulation and Thrombosis Outcomes

“…A few reports in the literature have described thrombosis improvement or resolution without anticoagulation [4,14,15,16,17,18]. Other case reports have described resolution of LS through treatment with both antibiotics and anticoagulation [25,32,33,34,35,36,37]. In a retrospective literature review of patients aged 8-49 years, 11 of 41 patients affected with LS received anticoagulation, and all of the patients in that group improved, with no mortality events described; however, 2 of the 30 patients in the nonanticoagulated group died from infectious complications (pneumonia and severe sepsis) [32].…”

“…The majority of the publications we cite here reported on exclusively adult patients with LS (age at diagnosis ≥18 years) [5,10,11,13,14,15,16,21,28,32,33,34,37,38,39]. Comparatively, data published in the pediatric population is very limited; however, we attempted to include all case reports and studies of exclusively pediatric patients as well [4,6,8,9,19,23,25,40,41,42].…”