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Lemierre's syndrome is a condition characterized by thrombophlebitis of the internal jugular vein and bacteremia following a recent oropharyngeal infection (Image below). Transsexualism is a rare condition with few reports on long-term safety of different treatment protocols and on the physical outcomes of medical treatment.

There is no case report of internal jugular thrombosis in those taking Estrogen hormone therapy in the medical literature to the best of our knowledge.

We are hereby presenting a case of a transsexual on estrogen with suspected Lemierre's syndrome.

A 28-year-old male to female transsexual and homosexual male on ethanyl estradiol and Spiranolactone under the direction of a local Transsexual clinic for one year, with a one time history of peritonsilar abscess six years ago which was incised and drained without further follow up, presented to the emergency department with a three day history of sore throat, difficulty and painful swallowing. The patient had taken a two day course of an unknown nonprescription antibiotic at home, then felt acutely worse on the day of presentation. The patient reported difficulty swallowing and pain upon opening the mouth and denied fever, chills, cough, chest pain, shortness of breath, other upper respiratory symptoms, or any other symptoms.

On physical exam patient was afebile, exhibited reduced ability to open the oral cavity, severe pharyngeal erythema, bilateral tonsillar enlargement and erythema partially occluding the airway, bilateral non-tender jugulo-digastric node enlargement and Male Tanner Stage 5; Female breast Tanner Stage 1. The patient had a leukocytosis of 13,800 with neutrophilia, Rapid streptococcal test: Negative, HIV/ Hepatitis B: Negative. An estrogen level was ordered, resulting in 287.5 pg/mL. CT scan with contrast of the neck: 8x5mm large left palatine tonsil with phlegmenous/early abscess formation with associated upper jugular lymphadenopathy, and lacked visualization of the proximal left internal jugular vein, secondary to thrombosis.. A left internal jugular vein thrombosis was confirmed by bedside ultrasound, demonstrating a non-compressible hyper-echoic thrombus (Bedside image below).

A concern for Lemierre's disease and progression of disease to sepsis led to blood cultures including anaerobic cultures, looking particularly for Fusobacterium Necrophorum, and empirically treated with piperacillin/tazobactam following local community resistance of Fusobacterium to Clindamycin. Patient was started on therapeutic anticoagulation with low molecular weight heparin. Inflammation and erythema started declining. The patient was bridged to Coumadin. Cultures remain negative. The patient was discharged on Amoxicillin/clavulanic acid for five more days, Coumadin for 3 months with a recommendation not to continue on Estrogen during the duration of Warfarin.

CASE PRESENTATION DISCUSSIONInternal Jugular Vein Thrombosis (IJVT) can be from a number of known causes such as head and neck infections that travel through post-pharyngeal spaces such as in this patient, or from other causes such as malignancies, polycythemia, hypercoagulable states, cervical injection drug use, surgery and venous lines. Its known sequel include sepsis, papilladema, and pulmonary embolism. This is an under diagnosed disease with a complex and often subtle physical presentation. Lemierre’s syndrome is life-threatening condition first described in 1936, characterized by an acute pharyngeal infection with Fusobacterium Necrophorum, and an associated IJV thrombosis and sepsis with possible bacteremic emboli anywhere downstream to infection such as pulmonary emboli that can result in life threatening lung abscess and empyema.

Estrogen levels above the therapeutic level of 200 pg/mL have been demonstrated to cause hyper-coagulable states in prospective studies of male-to-female transsexuals. Hormone replacement is associated with a higher risk of venous thromboembolism, although the risk seemed to be restricted to the first year of use. This case illustrates the importance of closely monitoring those taking Estrogen hormone replacement due to its diverse set of possible side effects.

Lemierre’s should be suspected in a patient in a hyper thrombotic state with pharyngitis, persistent septic symptoms despite treatment and possible pulmonary septic emboli.

REFERENCES1. Sinave CP, Hardy GJ, Fardy PW. The Lemierre syndrome: suppurative thrombophlebitis of the internal jugular vein secondary to oropharyngeal infection. Medicine (Baltimore) 1989; 68:85.

2. Chirinos JA, Lichtstein DM, Garcia J, Tamariz LJ. The evolution of Lemierre syndrome: report of 2 cases and review of the literature. Medicine (Baltimore) 2002; 81:458.

3. Asscheman H, Et al. Venous thrombo-embolism as a complication of cross-sex hormone treatment of male-to-female transsexual subjects: a review. Andrologia. 2013 Aug 15. doi: 10.1111/and.12150.

ACKNOWLEDGEMENT

John D'Angelo, DO

Chairman, Department of Emergency Medicine,Trinitas Regional Medical Center

Paymon Ebrahimzadreh, MS III; Fateema Islam, MS III

Lemierre's syndrome in a Male-to-Female transsexual.Vidya Kollu, MD

INTRODUCTION

Trinitas Regional Medical Center