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Paper text:
lemierre 's syndrome ( ls ) is an anaerobic suppurative thrombophlebitis involving the internal jugular vein , most often as a complication of pharyngeal and dental infection .
septicemia documented by at least one positive blood culture and one or more metastatic infective focus completes the features of this syndrome .
the disease most commonly affects teenagers and young adults ; therefore , it can occur in association with pregnancy .
however , a pubmed , ovid and embase search from 1950 to march 2007 , using search terms  lemierre 's syndrome and lemierre 's disease , pregnancy , gestation and cyesis ,  revealed only one case in which the pregnancy was complicated by premature delivery of an infant that suffered significant neurological deficits at 26 weeks .
we present an atypical case diagnosed by chance in a previously healthy , young woman who developed the syndrome in the second trimester of pregnancy .
a 21-year - old white female , g5p4004 , at 22 weeks was transferred to our hospital for evaluation of worsening sepsis .
she had presented to her local health facility with a three - day history of headache , nonproductive cough and fever .
she was evaluated and discharged home twice in 3 days with these symptoms . for the next week
, her symptoms persisted and worsened ; therefore , she was admitted to another medical facility in septic shock .
she was given intravenous fluids , placed on broad - spectrum antibiotics and required a dopamine drip due to persistent hypotension .
she demonstrated pancytopenia ( hb , 7 g / dl ; wbc , 1.7  10/l ; platelets 53  10/l ) and received 2 units of packed red blood cell transfusion prior to transfer to our facility . on presentation , she was somnolent but arousable .
her temperature was 36.2c , pulse was 88 beats / min , blood pressure was 96/54 mmhg , respiratory rate was 30/min with o2 saturation of 100% on 40% of oxygen .
her fundal height was compatible with her dates and fetal heart tone was 155 beats / min . her cranial nerves ii through xii were intact .
the patient was admitted to the medical intensive care unit with a diagnosis of septic shock .
intravenous cefepime , azithromycin , and levofloxacin were started after cultures were sent . on hospital day two , she developed respiratory distress requiring intubation and mechanical ventilation . during an attempt at placement of a left internal jugular intravenous access , copious amounts of purulent material exuded .
ct revealed suppurative mastoiditis with erosion of the mastoid bone towards the left transverse sigmoid sinus ; secondary septic thrombophlebitis involving the left transverse sinus , sigmoid sinus , internal jugular and left brachiocephalic veins .
in addition , bilateral pleural effusions and multiple bilateral patchy areas of focal airspace disease were noted , implying a septic phenomenon to the lungs . a diagnosis of ls was made .
otolaryngology and neurosurgery were consulted and patient underwent left mastoidectomy , evacuation of copious abscess deep in the left mastoid antrum , right myringotomy , and tube placement .
infectious disease service advised that her antibiotics be changed to piperacillin / tazobactam and metronidazole .
although her sepsis improved , it was difficult to wean her off the ventilator due to hemodynamic instability and atelectasis from right - sided pneumothorax .
a chest tube was placed and on removal , residual right lower lobe atelectasis persisted which resolved after a bronchoscopic examination to clear out secretions .
on extubation , the patient made a gradual but steady recovery and was discharged home after a 23-day hospital stay .
arrangements were made for her to continue with physical therapy and intravenous antibiotics for 6 weeks . throughout her entire hospital course ,
follow - up growth scans suggested intrauterine growth restriction , necessitating induction of labor at 37 weeks .
the patient had a successful induction with spontaneous vaginal delivery of a live female infant weighing 2892 grams with apgar scores of 9 at one minute and 9 at five minutes .
lemierre 's syndrome is a potentially fatal multisystemic disease resulting from complication of anaerobic infection of the head and neck usually in previously healthy individuals . although few cases had been described prior , it was lemierre , a professor of bacteriology and infectious disease at the claude bernard institute in paris , who first wrote a comprehensive review of this disease in 1936 and subsequently earned the name by which the syndrome is now known .
the infectious causes are diverse and are part of the normal flora of the mouth , genital , gastrointestinal , and upper respiratory tracts .
fusobacterium necrophorum , a highly virulent obligatory anaerobe , is often but not invariably associated with this condition and was not isolated in our case .
the original infection is usually a tonsillitis , pharyngitis , or odontogenic infection but has also been described following penetrating oropharyngeal trauma .
our patient did not volunteer the typical history of initial oropharyngeal infection , neither was her neck tender despite the presence of mastoid abscess .
this is not surprising because unlike infection and thrombophlebitis involving the anterior compartment of the lateral pharyngeal space , that involving the posterior space may remain asymptomatic until the disease progresses systematically to include septic pulmonary embolism .
had the placement of an internal jugular venous catheter not been performed in our case , the diagnosis might have been delayed with serious consequences .
however , this led to the chain of events that revealed the diagnosis and resulted in a successful outcome by enabling the removal of the primary focus of infection .
the advent of high - resolution computed tomography for delineating the extent of thrombophlebitis has led to more cases being diagnosed and should be performed as early as possible .
the disease most commonly affects teenagers and young adults ; therefore , it is not difficult to envisage its occurrence in association with pregnancy .
mclean and tyler   reported a nearly fatal case in a 23-year - old primigravida that resulted in premature delivery of an infant that suffered significant neurological deficits at 26 weeks . in that case ,
ls was complicated by cardiopulmonary collapse due to mediastinitis , pericardial effusion and tamponade that required mechanical ventilation , cardioversion , and emergent pericardiocentesis .
the traditional view is that microorganisms gain access to the amniotic cavity after rupture of membranes ; however invasion of the amniotic cavity can take place prior to this .
found that 13.6% of women with preterm labor and intact membranes had a positive amniotic fluid culture , and that such women are more likely to rupture than those with a negative culture .
however , our case fortunately resulted in a live birth of an apparently healthy child at term .
the natural history of cases described in the literature is similar to that encountered in these two pregnant cases ( this case and the one before it ) , hence pregnancy can not be said to aggravate the course of the disease . aside from sepsis ,
decrease in colloid oncotic pressure encountered in pregnancy , in conjunction with the added effect of acute lung injury causing lung exudation , surfactant loss and diminished lung volume , and vascular shunting , can result in significant hypoxemia .
intubation and mechanical ventilation is preferred in pregnant women if respiratory failure exists or is imminent to avoid defective oxygen delivery to the baby .
an extended course of intravenous antibiotics based on the results of cultures is often required and although it is currently advocated that it be continued for 6 weeks , this is based solely on expert opinion .
goldenberg et al . , in their study of the disease in the pediatric age group , found thrombophilic abnormalities in all investigated cases of ls in their series but in only one patient did the abnormality persisted after they were retested 2 to 6 months after diagnosis .
these authors suggested that these thrombophilic abnormalities might be an epiphenomenon of the acute inflammatory prothrombotic state rather than an intrinsic hypercoagulability in these patients .
therefore , routine tests for thrombophilia may not be justified or cost effective until more data are available .
the role of anticoagulant therapy in lemierre 's syndrome is controversial butmay be beneficial , however , no control trial exists studying the efficacy of anticoagulant therapy in this condition . because of the possibility of extension of the infective agents , some suggest that anticoagulant therapy should be reserved for thrombosis retrograde to the cavernous sinus .
the likely effect of pregnancy on the course of disease and vice - versa can not be ascertained from this single case and the one before it .
nevertheless , the severe and sometimes life - threatening nature of this syndrome necessitates an enhanced awareness by primary care and emergency room physicians and obstetricians .
when a previously healthy patient presents with sepsis following an unabating oropharyngeal infection in association with pulmonary symptoms , a chest x - ray should be performed at the minimum followed by a ct scan of the head , neck , and chest if ls can not be excluded .
management as outlined by our case and those of others necessitates a multidisciplinary approach if mortality is to be reduced .
lemierre 's syndrome is an anaerobic suppurative thrombophlebitis involving the internal jugular vein secondary to oropharyngeal infection . 
 there is only one previous case report in pregnancy which was complicated by premature delivery of an infant that suffered significant neurological damage . 
 we present an atypical case diagnosed in the second trimester with a live birth at term . by reporting this case 
 , we hope to increase the awareness of obstetricians to the possibility of lemierre 's syndrome when patients present with signs of unabating oropharyngeal infection and pulmonary symptoms .