A previously healthy 58-year-old male was transferred from another hospital to our department due to sepsis of undetermined origin with an unfavorable evolution under broad-spectrum antibiotic therapy (imipenem-cilastatin and clarithromycin). The patient complained of pain in the lumbar region and fever throughout the preceding two weeks. He also described asthenia, anorexia and a recent weight loss of 3 kg. The medical history was unremarkable. He was a non-smoker and reported no drug abuse, recent travel or contact with animals.
Upon admission to our hospital, the patient presented a high fever of 39.3 °C with hemodynamic stability. The physical examination showed no heart murmur or signs of cardiac congestion, but the presence of bilateral painless swollen legs was observed. Pulmonary auscultation showed respiratory crackles on the right side. The neurological, abdominal, cutaneous and osteoarticular examinations were unremarkable.
Laboratory examinations revealed the following results: hemoglobin 116 g/l (normal range: 140–180 g/l); leukocyte count, 33.3*109 cells/l (4–11*109 cells/l) without left-band shift; C-reactive protein, 114.6 mg/l (0–10 mg/l); creatinine, 56 μmol/l (62–106 μmol/l), aspartate transaminase, 174 U/l (12–50 U/l); alanine aminotransferase, 209 U/l (14–50 U/l); alkaline phosphatase, 406 U/l (25–102 U/l); gamma-glutamyl transpeptidase, 517 U/l (9–40 U); total bilirubin, 76 μmol/l (7–25 μmol/l); conjugated bilirubin, 66 μmol/l (0.5–9.5 μmol/l); International Normalized Ratio, 1.38. Serology assays for hepatitis B virus, hepatitis C virus and human immunodeficiency virus were negative. Urinary sediment was unremarkable.
As there was a high suspicion of spondylodiscitis, blood cultures were ordered and a thoraco-abdominal computed tomography (CT) scan was performed. The scan revealed thromboses of the two iliac veins, abscesses in the right iliac muscle (2.3 × 2.0 cm) and the left psoas muscle (6.0 × 4.8 cm), a right pleural effusion complicating a pneumonia, as well as a L4-L5 spondylodiscitis (Fig. 1). Lumbar magnetic resonance imaging confirmed an L4-L5 spondylodiscitis and the presence of abscesses in the muscles. One blood culture (taken initially as an outpatient before antibiotic administration) and the culture of the pus drained from the right psoas muscle grew F. nucleatum. The right pleural effusion was also drained and confirmed the presence of an empyema without bacterial growth (under antimicrobial therapy). Antibiotic therapy was then switched to amoxicillin-clavulanate in combination initially with clindamycin as F. nucleatum is pan-susceptible to these antimicrobials.
When considering the presence of F. nucleatum bacteremia with a complicated pneumonia, a spondylodiscitis and multiple muscle abscesses, we suspected a case of Lemierre’s syndrome and attemted to identify the source of the infection. An orthopantomogram, a transoesophageal echocardiography as well as a cerebral CT scan were unremarkable. The duplex sonography confirmed bilateral venous iliac thromboses. As the patient presented lower gastrointestinal bleeding under anticoagulation (acenocoumarol), a colonoscopy was performed, but revealed no sign of malignancy or mucosal lesions. An inferior vena cava filter was put in place and withdrawn at one month and anticoagulation was continued for a total duration of 3 months. Antibiotic treatment was given intravenously for 6 weeks. At 3 months, duplex ultrasound showed complete resolution of the thromboses and clinical follow-up was normal. Spinal magnetic resonance imaging at 9 months showed sequelae of the L4-L5 spondylodiscitis.
We searched PubMed using the following terms “atypical + Lemierre syndrome” and then selected the atypical cases as illustrations. The search was not exhaustive.