Septic Thrombophlebitis Research Articles

Lemierre Syndrome Associated with Streptococcus constellatus and Atypical Vascular Involvement: A Case Report and Review of the Literature

Background: Lemierre syndrome is a rare and life-threatening disease. It is characterized by septic thrombophlebitis of the internal jugular vein, historically associated with Fusobacterium necrophorum infection. However, atypical cases and associations with other organisms have been reported. Methods: Here, we describe a challenging case of Lemierre syndrome in a 71-year-old woman caused by Streptococcus constellatus and review the related literature. Case: The patient experienced multiple hospital admissions due to misdiagnoses and developed thrombosis involving the internal jugular vein and transverse sinus bilaterally, pulmonary complications including the formation of a pseudoaneurysm, and occipital abscess. She presented with headaches, neck pain, and blindness. Prolonged antibiotic therapy was administered, leading to gradual improvement of symptoms, with partial resolution of blindness. Prophylaxis with intramuscular penicillin was prescribed at discharge. Conclusions: Our case underscores the importance of considering Lemierre syndrome in patients who present with multiple thrombotic events affecting the intracranial circulation and/or jugular veins, particularly in those already receiving anticoagulation therapy or with no identifiable cause for thrombosis, even in the absence of sore throat or fever.

Multiple Nodular Condensation from Peripheral Excavations Revealing Septic Thrombophlebitis of Jugular Vein: Think of Lemierre Syndrome

Multiple Nodular Condensation from Peripheral Excavations Revealing Septic Thrombophlebitis of Jugular Vein: Think of Lemierre Syndrome

Septic Thrombophlebitis of the Umbilical Vein Complicating an Intrahepatic Umbilical Venous Catheter in a Premature Newborn

Septic thrombophlebitis of the umbilical vein (SVT) is a serious pathology in neonates, characterized by inflammation of the umbilical vein due to bacterial infection, with 90% of cases linked to the use of central venous catheters (CVCs). This inflammation can lead to thrombus formation as part of the immune system's response. Premature newborn, 33 weeks gestation, delivered vaginally, with history of gestational diabetes, twin pregnancy, macrosomia and premature rupture of membranes (57 hours), initially well adapted to extrauterine life, with a birth weight of 1575 g. The infant was admitted to neonatology for respiratory distress, and an intrahepatic umbilical catheter was inserted. On the 5th day of hospitalization, the infant developed a nosocomial Klebsiella pneumoniae infection, complicated by a fulminant hemorrhagic syndrome with hematemesis and melena. The clinical course was further complicated by multivisceral failure, requiring hepatic Doppler ultrasound, which revealed umbilical vein thrombosis. The newborn was treated with curative doses of Lovenox, while targeted antibiotic therapy was maintained for three weeks after negative blood cultures, resulting in significant clinical and biological improvement. Based on this case, we conclude that it is advisable to think about umbilical vein thrombosis and to perform an abdominal ultrasound in the presence of any digestive hemorrhage in a newborn or an intrahepatic or peripheral umbilical venous catheter, or in subjects at risk (hypotrophy, polycythemia, transfusion on the catheter) in order to screen for possible umbilical or portal vein thrombosis.

Lemierre syndrome due to Klebsiella pneumoniae: a rare case report with review of literature

Lemierre’s syndrome, a forgotten clinical entity, is characterised by septic thrombophlebitis of the internal jugular vein due to oropharyngeal infection. In the past, it was mainly associated with Fusobacterium necrophorum infections. We present a unique case report of Lemierre’s syndrome due to Klebsiella pneumoniae. Patient reported with progressive swelling involving right side of the neck that eventually increased in size. After confirming the diagnosis through CECT, patient was managed through combined medical and surgical therapy in the form of incision and drainage, targeted antibiotic therapy, glycaemic control and anticoagulation therapy. This case report highlights the association of poor glycaemic control in the pathogenesis and the isolation of Klebsiella pneumoniae as the rare organism of Lemierre’s syndrome.

Association of pathogenic determinants of Fusobacterium necrophorum with bacteremia, and Lemierre’s syndrome

Fusobacterium necrophorum is a Gram-negative anaerobic bacterium responsible for localized infections of the oropharynx that can evolve into bacteremia and/or septic thrombophlebitis of the jugular vein or peritonsillar vein, called Lemierre’s syndrome. To identify microbial genetic determinants associated with the severity of this life-threatening disease, 70 F. necrophorum strains were collected and grouped into two categories according to the clinical presentation: (i) localized infection, (ii) bacteremia with/without Lemierre’s syndrome. Comparative genomic analyses revealed two clades with distinct genetic content, one clade being significantly enriched with isolates from subjects with bacteremia. To identify genetic determinants contributing to F. necrophorum pathogenicity, genomic islands and virulence factor orthogroups (OVFs) were predicted. The presence/absence profiles of OVFs did not group isolates according to their clinical category, but rather according to their phylogeny. However, a variant of lktA, a key virulence factor, with a frameshift deletion that results in two open reading frames, was associated with bacteremia. Moreover, a genome-wide association study identified three orthogroups associated with bacteremic strains: (i) cas8a1, (ii) a sodium/solute symporter, and (iii) a POP1 domain-containing protein. Further studies must be performed to assess the functional impact of lktA mutation and of these orthogroups on the physiopathological mechanisms of F. necrophorum infections.

A case of multidrug-resistant intractable pylephlebitis and intra-abdominal abscess due to perforated appendicitis successfully treated with open abdominal management

BackgroundPylephlebitis, a rare and lethal form of portal venous septic thrombophlebitis, often arises from infections in regions drained by the portal vein. Herein, we report a case of peritonitis with portal vein thrombosis due to acute severe appendicitis, managed with intensive intraperitoneal drainage via open abdominal management (OAM).Case presentationA 19-year-old male with severe appendicitis, liver abscesses, and portal vein thrombosis developed septic shock and multi-organ failure. After emergency interventions, the patient was admitted to the intensive care unit. Antibiotic treatment based on cultures revealing multidrug-resistant Escherichia coli and Bacteroides fragilis and anticoagulation therapy (using heparin and edoxaban) was initiated. Despite continuous antibiotic therapy, the laboratory results consistently showed elevated levels of inflammatory markers. On the 13th day, open abdominal irrigation was performed for infection control. Extensive intestinal edema precluded wound closure, necessitating open-abdominal management in the intensive care unit. Anticoagulation therapy was continued, and intra-abdominal washouts were performed every 5 days. On the 34th day, wound closure was achieved using the anterior rectus abdominis sheath turnover method. The patient recovered successfully and was discharged on the 81st day.ConclusionsAlongside appropriate antibiotic selection, early surgical drainage and OAM are invaluable. This case underscores the potential of anticoagulation therapy in facilitating safe surgical procedures.

Septic Thrombophlebitis of the Internal Jugular Vein due to Blastomycosis Mastoiditis Resulting in Lemierre Syndrome: A Case Report (P7-5.013)

Septic Thrombophlebitis of the Internal Jugular Vein due to Blastomycosis Mastoiditis Resulting in Lemierre Syndrome: A Case Report (P7-5.013)

A Rare Case of Lemierre’s Syndrome due to Veillonella Parvula: A Dangerous and Forgotten Complication of a Septic Condition

This clinical case presents an unusual case of Lemierre’s syndrome (LS) in a young woman of 38-year-old. She arrived in the Emergency Department with a high fever and pharyngology resistant to antibiotic therapy with clarithromycin, ceftriaxone, and cortisone for two weeks. At the blood sampling, there is a marked leucocytosis, and the advice of the otolaryngologist is required given the strong pain in the throat. Due to the tonsillar abscess, a neck CT with a contrast medium is necessary for the otolaryngologist’s opinion. The CT shows thrombosis of the jugular vein and left subclavian, with thickening of soft perivascular tissues; these findings suggest Lemierre’s syndrome: a septic thrombophlebitis of the jugular vein that occurs as a complication of a peritonsillar abscess. The diagnostic process is then completed with a chest HR-CT, which reveals lung density and excavation areas suggesting tuberculosis. Blood culture reveals the presence of Veillonella Parvula (an anaerobic gram-negative coccus), sputum culture reveals the presence of some colonies of Enterobacter cloacae complex, real-time PCR examination on sputum reveals the presence of Streptococcus Pneumoniae and the borderline presence of rhinovirus. Microbiologists, after these results and neck and chest CT with a contrast agent, agree with the diagnosis of suspected LS at an early stage: a septic dissemination fortunately limited only to the neck and lungs region.

Pediatric Lemierre's Syndrome: A Comprehensive Literature Review.

Lemierre syndrome is a rare, potentially fatal complication of oropharyngeal infections characterized by septic thrombophlebitis of the internal jugular vein. It primarily affects healthy adolescents and young adults. Its incidence declined after the antibiotic era, but it may have resurged in recent decades, likely due to judicious antibiotic use and increasing bacterial resistance. Prompt diagnosis and treatment are imperative to prevent significant morbidity and mortality. Lemierre syndrome has been called "the forgotten disease," with a reported incidence of around 3.6 cases per million. The mean age at presentation is around 20 years old, though it can occur at any age. Lemierre Syndrome follows an oropharyngeal infection, most commonly pharyngitis, leading to septic thrombophlebitis of the internal jugular vein. F. necrophorum is the classic pathogen, though other organisms are being increasingly isolated. Metastatic infections, especially pulmonary, are common complications. Contrast-enhanced CT of the neck confirming internal jugular vein thrombosis is the gold standard for diagnosis. Long-course broad-spectrum IV antibiotics covering anaerobes are the mainstays of the disease's treatment. Anticoagulation may also be considered. Mortality rates are high without treatment, but most patients recover fully with appropriate therapy. Lemierre syndrome should be suspected in patients with prolonged pharyngitis followed by unilateral neck swelling and fevers. Early diagnosis and prompt antibiotic therapy are key, given the potential for disastrous outcomes if untreated. An increased awareness of Lemierre syndrome facilitates its timely management.

Proteomic Characterization of Plasma in Lemierre's Syndrome.

The underlying mechanisms of thrombosis in Lemierre's syndrome and other septic thrombophlebitis are incompletely understood. Therefore, in this case control study we aimed to generate hypotheses on its pathogenesis by studying the plasma proteome in patients with these conditions. All patients with Lemierre's syndrome in the Skåne Region, Sweden, were enrolled prospectively during 2017 to 2021 as cases. Age-matched patients with other severe infections were enrolled as controls. Patient plasma samples were analyzed using label-free data-independent acquisition liquid chromatography tandem mass spectrometry. Differentially expressed proteins in Lemierre's syndrome versus other severe infections were highlighted. Functions of differentially expressed proteins were defined based on a literature search focused on previous associations with thrombosis. Eight patients with Lemierre's syndrome and 15 with other severe infections were compared. Here, 20/449 identified proteins were differentially expressed between the groups. Of these, 14/20 had functions previously associated with thrombosis. Twelve of 14 had a suggested prothrombotic effect in Lemierre's syndrome, whereas 2/14 had a suggested antithrombotic effect. Proteins involved in several thrombogenic pathways were differentially expressed in Lemierre's syndrome compared to other severe infections. Among identified proteins, several were associated with endothelial damage, platelet activation, and degranulation, and warrant further targeted studies.

Multiple Cold Abscesses of a Chest Wall with Ribs Destruction in a Hemodialysis Patient

Cold abscess of the chest wall is an uncommon disease that is mainly caused by tuberculous infection. Staphylococcal cold abscesses of the chest wall are extremely rare and usually clinically occult, frequently leading to a delay in diagnosis and consequently severe infectious complications. We report an 88-year-old woman with end-stage renal disease, who presented with an exit site infection of a cuffed tunneled dialysis catheter caused by methicillin-resistant Staphylococcus aureus (MRSA). Despite the removal of the catheter and administration of antibiotics, she developed refractory MRSA bacteremia. Computed tomography (CT) of the chest revealed septic thrombophlebitis with metastatic cold abscesses of the chest wall and ribs destruction. Although CT-guided drainage and vancomycin therapy eliminated MRSA bacteremia, the chest wall abscesses did not resolve. Patients fitted with a central venous dialysis catheter are at risk of septic thrombophlebitis with metastatic cold abscesses of the chest wall that are resistant to antibiotic therapy. Early identification is based on serial blood cultures and prompt CT scans. Surgical management coupled with antibiotic therapy can eradicate the source of infection and improve patients’ outcomes.

Diffuse Maculopapular Eruption in a Patient with Lemierre’s Syndrome

Lemierre’s syndrome is a potentially fatal septic thrombophlebitis of the internal jugular vein (IJV) requiring prompt diagnosis and treatment. In this report, we document a unique presentation of an EBV-associated morbilliform eruption coincident with the onset of Lemierre’s syndrome. Furthermore, we argue that a diffuse maculopapular eruption in patients with Lemierre’s syndrome should prompt workup for EBV infection.

Spectrum on Non-Surgical Causes of Internal Jugular Vein Thrombosis In Otorhinolaryngology Practice

Internal jugular vein thrombosis is an uncommon entity secondary to various aetiologies. In the pre-antibiotic era, it was commonly associated with Lemierre syndrome, a septic thrombophlebitis of the internal jugular vein secondary to oropharyngeal infection. With the advent of antibiotics, Lemierre syndrome has become a rare entity and is often overlooked. Internal jugular vein thrombosis may also occur in the setting of malignancy, a combination referred to as Trousseau’s syndrome, which is not well documented.

CTNI-83. MATRIX REGIMEN FOR NEWLY DIAGNOSED PRIMARY CNS DIFFUSE B-CELL LYMPHOMA

Abstract We report our experience treating 25 patients with newly diagnosed primary CNS diffuse B-cell lymphoma with the MATRix regimen as reported by Ferreri, et al in 2016. The median age was 66 y (range 30-85); 12 patients were men and 13 were women. Twenty-three had cerebral mass lesions, 1 had a paraspinal mass, and 1 leptomeningeal disease. Treatment consisted of 4 cycles of rituximab 375 mg/sq m (Days 1, 6); methotrexate 3.5 g/sq m (Day 2); cytarabine 2 g/sq m every 12 h (Days 3, 4); and thiotepa 30 mg/sq m (Day 5). Patients without disease progression received carmustine 400 mg/sq m and thiotepa 5 mg/kg followed by autologous bone marrow transplantation. Four patients were switched to MATRix after 6 cycles of methotrexate and rituximab and received only 2 initial cycles including cytarabine and thiotepa. Overall, 18 patients completed initial chemotherapy. Sixteen of these 18 went on to transplantation; one patient had had early disease progression, and in one no stem cells could be collected. Five patients (median 73 y; range 64-85 y) died early after 1-2 cycles. The causes of early death were neutropenic fever, septic thrombophlebitis, and cardiac arrest. One of the 16 patients who completed the entire regimen including transplantation died of fungal pneumonia and the other 15 experienced no serious acute toxicity. One patient had late disease progression and 2 developed symptomatic leukoencephalopathy. Seventeen of the 25 patients remain alive. Median TTP for the cohort is 605 days, and OS 738 days. These preliminary data suggest that the MATRix regimen in safe and highly effective in the newly diagnosed setting. However, the high incidence of early death in patients over 65 y shows that the regimen is too toxic for older individuals.

Carfilzomib in Combination with Dexamethasone, Cyclophosphamide, Etoposide, and Cisplatin (KDCEP) for the Treatment of Relapsed/Refractory Aggressive Plasma Cell Dyscrasias

Background: Dexamethasone with Cyclophosphamide, Etoposide and Cisplatin (DCEP) is an effective regimen in aggressive relapsed/refractory MM (RRMM) with overall response rates (ORR) as high as 58% [Dadacaridou, M. et al JBUON 2007]. ORR is lower (35 - 45%) in patients (pts) exposed to novel therapeutics such as immunomodulatory agents (IMiD), proteasome inhibitors (PI), and anti-CD38 antibodies (mAb) [Park, Silvia et al Ann. Hematol 2014; Goldsmith, Scott et al. Blood2021]. The addition of bortezomib (V) to DCEP demonstrated ORR of 47% [Valla, Kelly et al. Blood 2014]. Due to common use of V front-line, most pts are V exposed by the time treatment for relapse is indicated; therefore, we began using carfilzomib (K) with DCEP. K has been utilized with DPACE with ORR of 77% [Alsouqi, A. et al. Clin Lymphoma Myeloma Leuk 2021], however an anthracycline may not be feasible in heavily pretreated pts. We present the first reported outcomes of RRMM pts treated with KDCEP. Methods:This is a case series from the University of Utah Huntsman Cancer Institute of all consecutive pts treated with KDCEP from 2017-2022, conducted under IRB approval. Demographic, disease, treatment, and response data were retrospectively collected. Responses were assessed by IMWG criteria, and adverse events were graded by CTCAE v.5. Descriptive statistics were utilized; a Kaplan-Meier estimate was used for the time to event analysis. Results:Six pts received KDCEP (Table 1). The median age was 63.5 years (range 47 - 73). All pts had ECOG performance status of 1-2, and 67% were male. Median prior treatment lines were 2.5 (range 2 - 7) with 33% undergoing prior autologous stem cell transplant (autoHCT). All pts were exposed to V with 67% refractory. Thirty-three percent had prior K exposure, however none were considered refractory to K, and 67% were refractory to a PI, IMiD and mAb. Two pts received KDCEP as chemo-mobilization prior to autoHCT. All pts had IgG or IgA disease with 67% having high risk cytogenetics (33% 17p del, 50% gain 1q21) and 1 pt had primary plasma cell leukemia. R-ISS stage at diagnosis was: stage 3 (n=2), stage 2 (n=1) unknown (n=3). All pts had baseline echocardiograms with ejection fractions prior to treatment ranging from 57 - 78%. Pts received K on Days 1 and 8 of each 28-day cycle, prophylactic growth factor, and standard antimicrobial prophylaxis. Two pts received 2 cycles, all others received 1. The overall response rate was 100% and included the following best responses: PR (n = 4), VGPR (n = 1), CR (n = 1). One patient that initially achieved a PR relapsed within 60 days and died awaiting enrollment on a clinical trial, however all other pts were successfully bridged to their next line of therapy. Five pts had MRD data available after treatment, each demonstrating detectable MRD. Median progression free survival was 6.7 months. Median time to ANC nadir was 11 days (range 9-14) and to ANC recovery, defined as ANC > 1000/uL, was 18 days (range 14 - 23). Median time to platelet nadir was 12.5 days (range 9-23) and to platelet recovery, defined as platelets > 100 k/uL, was 25 days (range 20 - NR). Median time to next cycle or treatment was 43 days (range 31-54). Sixty-seven percent of pts had a grade ≥ 3 toxicity including neutropenic fever (n=2), sepsis due to pseudomonas bacteremia (n=1), and septic thrombophlebitis (n=1). No cardiac toxicities or treatment related deaths were observed. Discussion:In this cohort of heavily pre-treated pts who received KDCEP, ORR was high indicating the regimen is active in this population. KDCEP may be a viable option for RRMM pts, particularly in those who are triple class refractory or with high risk cytogenetic abnormalities. Our data suggests that KDCEP may be an effective regimen to bridge patients to the next line of therapy; however all patients should be monitored closely for infection.

Severe Thromboses in Neonates.

Severe thrombosis in the neonate presents a diagnostic challenge to the clinician as the benefits and risks for treatment must be weighed with every medical decision. Aortic thromboses, large right atrial thromboses, and septic thrombophlebitis present unique clinical challenges that must be managed in the appropriate clinical setting with appropriate subspecialty support. Unfortunately, there is limited data on what the ideal therapy should be for these specific types of cases. In this review, we will discuss these types of severe thromboses that may occur in neonates and potential therapies that may offer benefits.

Liver Abscesses Due to Fusobacterium nucleatum: A Case Series

Abstract Fusobacteriae are anaerobic gram-negative bacilli, classically involved in the development of septic thrombophlebitis of the internal jugular vein in Lemierre syndrome. The goal of this study is to describe the various presentations, treatment course, and outcomes of patients with Fusobacterium liver abscess(es) and review available literature on this topic. We retrospectively reviewed our microbiologic database for positive cultures of Fusobacteriae species in a tertiary care hospital from July 2018 through November 2022 and identified 5 cases of liver abscess that involved Fusobacterium nucleatum species. Patients presented with a syndrome of fever and chills, abdominal pain, and nausea and vomiting. Laboratory investigations revealed leukocytosis and transaminitis, and imaging revealed liver abscess(es). The identification of the F. nucleatum bacteria involved invasive aspiration of the abscess in all but one case, where it was identified on blood cultures. One patient had a florid picture of sepsis and acute respiratory distress syndrome. All patients responded well with intravenous antibiotics and were discharged home to complete a prolonged course until radiographic resolution. In 3 patients, history and imaging were suggestive of possible sources that may have hematogenously spread to the liver, with history of recent appendectomy in 1 patient, possible appendicitis on computed tomography of the abdomen in another patient, and recent dental manipulation in the third patient. In conclusion, Fusobacteriae should be included in the differential diagnosis of liver abscess, especially in the setting of recent appendicitis or dental disease. Effective treatment includes surgical drainage when feasible and prolonged course of antibiotics with goal of radiologic resolution.

A Case of Septic Renal Vein Thrombophlebitis Secondary to Pyelonephritis Responded to Medical Treatment

A Case of Septic Renal Vein Thrombophlebitis Secondary to Pyelonephritis Responded to Medical Treatment

Septic thrombophlebitis of portal and splenic vein secondary to Fusobacterium nucleatum: A case report of an abdominal variant of lemierre syndrome.

Septic thrombophlebitis of the internal jugular vein also known as Lemierre syndrome occurs secondary to an oropharyngeal infection often leading to septic embolisms to distant sites. Anaerobic gram-negative bacillus, Fusobacterium nucleatum and Fusobacterium necrophorum are commonly isolated organisms. Fusobacterium species has also been reported to complicate an intra-abdominal infection leading to septic thrombophlebitis of portal vein also known as pylephlebitis or abdominal variant of lemierre syndrome. The patient was a middle-aged female patient with chief complaints of abdominal discomfort, intermittent fever and vomiting for one month. The final diagnosis was septic thrombophlebitis of portal and splenic vein secondary to Fusobacterium nucleatum. Patient was managed with broad spectrum intravenous antibiotics with coverage against gram-negative bacilli, anaerobes, and aerobic streptococcus species with therapeutic anticoagulation. Patient gradually improved and was discharged on oral apixaban. She was instructed to follow up with gastrointestinal specialist upon discharge in anticipation of the need for liver transplant in future. Due to its high mortality and associated long term disease morbidity, clinicians should always strive towards early diagnosis and treatment of the condition with involvement of multidisciplinary teams.

Lung abscess as a complication of Lemierre Syndrome in adolescents: a single center case reports and review of the literature

BackgroundFusobacterium necrophorum is an anaerobic, gram-negative, non-motile, filamentous, non-spore forming bacillus found in the oral cavity, gastrointestinal tract, and female genital tract, responsible of a rare disease named Lemierre Syndrome, characterized by septic thrombophlebitis of the internal jugular vein, which mainly affects previously healthy adolescents and young adults; some risk factors are reported, as smoking or primary viral or bacterial infection leading to the disruption of mucosa. The syndrome originates commonly from an upper respiratory infection such as pharyngotonsillitis, acute otitis media, cervical lymphadenitis, sinusitis, or odontogenic abscess, and may result in multiorgan metastasis, more frequently leading to pulmonary complications, especially lung abscesses.Case presentationWe describe two cases of adolescents with atypical Lemierre Syndrome evaluated in a tertiary care center, one with a confirmed infection by Fusobacterium necrophorum and one with a presumptive diagnosis based on clinical features, who developed lung abscesses needing a prolonged antibiotic course and hospitalization. Of interest, both were user of electronic cigarette, configuring a possible new risk factor. The proper diagnosis of Lemierre Syndrome is often difficult to establish, so a high degree of suspicion is needed, especially in the case of lung abscesses in otherwise healthy adolescents.ConclusionThe current study will contribute to providing insight into Lemierre Syndrome clinical presentation and management in adolescents, promoting awareness for a rare but potentially fatal disease. Moreover, it suggests a possible relationship between Lemierre syndrome and the use of electronic cigarette, that should be investigated by future studies.