Lemierre syndrome associated Cavernous Sinus Thrombosis and Development of Internal Carotid Artery Narrowing: A Case Report and Review of Literature
Jiping Zhou1, Sabeena Malik2
1Detroit Medical Center, 2Department of Neurology, Detroit Medical Center/Wayne State University
Objective:

To report a case of Lemierre syndrome complicated by cavernous sinus thrombosis (CST) and bilateral
internal carotid artery (ICA) stenosis without cerebral infarction in a young woman and summarize the literature on Lemierre syndrome with arterial involvement.

Background:

Lemierre syndrome is a complication of oropharyngeal infection characterized by thrombosis of the
internal jugular vein (IJV) with septic thrombophlebitis followed by bacteremia and septic emboli dissemination to distal organs, but arterial involvement is extremely rare.

Design/Methods:

A 24-year-old previously healthy woman presented to the hospital with one week of progressive jaw pain and neck swelling after recent tooth extraction. Physical examination showed left ptosis without other neurologic deficits. Computer tomography (CT) of the neck found multiple anterior neck abscesses. The patient underwent emergent incision and drainage of abscess at the left lateral pharyngeal, left masticator, and bilateral sublingual space. Vascular imaging revealed findings of non-occlusive thrombus in the left IJV, bilateral cavernous sinus thrombosis (CST), and septic emboli in bilateral apical lungs, consistent with Lemierre syndrome. Neuroimaging showed no cerebral infarction even though a digital subtraction angiogram (DSA) demonstrated bilateral ICA severe stenosis (>70%) in both extracranial and intracranial segments. Cultures of abscess samples proved positive for Candida albicans and Streptococcus constellatus. The patient received treatment with full-coverage antibiotics and anticoagulation therapy resulting in an excellent outcome. 

Results:

A literature search yielded 29 cases of Lemierre syndrome with arterial complications, which manifested as ICA occlusion or stenosis, or mycotic aneurysm development. Despite variable clinical symptoms, most patients developed CST and were all treated with antibiotics. Anticoagulation therapy is controversial given scarce and inconsistent evidence.

Conclusions:

The presented case and literature review emphasize the variability of the clinical course of Lemierre syndrome. Septic CST can be a rare cause of ICA occlusion, stenosis, or pseudoaneurysms because of arteritis, and thus expedited workup is necessary.

10.1212/WNL.0000000000202854