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Copyright 2004 American Medical Association. All Rights Reserved.Applicable FARS/DFARS Restrictions Apply to Government Use.2004
Fusobacterium organisms are obligate anaerobic gram-negative bacillibelonging to the family Bacteroidaceae. Of the 15 species recognized, Fusobacterium nucleatum and Fusobacteriumnecrophorum are the most frequently isolated species from clinicalspecimens. These species are most commonly found in the mouth and to a lesserextent in feces and the urogenital tract but rarely give rise to severe disease.1 We describe a previously healthy woman with a historyof severe periodontal disease who developed septic thrombophlebitis of theorbit and cavernous sinus caused by F nucleatum. Weare unaware of previous reports of septic thrombophlebitis of the orbit andcavernous sinus caused by F nucleatum.
A previously healthy 55-year-old woman had a 1-month history of left-sidedheadaches that were treated with pain medications. Her medical history wasnotable for severe periodontal disease and a previous partial thyroidectomyfor a benign mass. The patient subsequently developed severe left orbitalpain. Computed tomography and magnetic resonance imaging of the orbits andbrain were performed, and the findings demonstrated cavernous sinus enlargementand enhancement on the left side. Thoracoabdominal computed tomography andcerebrospinal fluid evaluation results were negative. Infectious and noninfectiousinflammatory processes, including Tolosa-Hunt syndrome and granulomatous diseases,were considered in the differential diagnosis, and the patient was treatedwith antibiotics and high doses of steroids. One week prior to admission toour institution, she developed bilateral proptosis and intractable pain. Shewas referred to our institution for further evaluation and treatment.
On examination she had bilateral proptosis with severe periocular edemaand erythema with conjunctival hyperemia and chemosis. She was unable to openher eyes. Her visual acuity was counting fingers OU, but this could not beassessed reliably because of poor patient cooperation due to extreme pain.Extraocular motility was markedly limited in all fields of gaze bilaterally.The pupils measured 2 mm bilaterally with normal pupillary light reaction.Additional magnetic resonance images of the brain and orbits were obtained.Compared with the magnetic resonance images obtained 1 week earlier, therewas now involvement of the right cavernous sinus (Figure 1). Proptosis of both globes with reticulated abnormal enhancementof the retrobulbar fat in both orbits was also noted. There was shaggy enhancementinvolving the optic nerve sheaths and the walls of the enlarged superior ophthalmicveins (Figure 2). The paranasalsinuses were clear. Magnetic resonance angiography revealed diffuse narrowingof the petrous, cavernous, and supraclinoid segments of the left carotid artery.The most likely diagnosis based on the clinical findings and imaging studieswas cavernous sinus thrombosis, secondary to an infectious or inflammatoryprocess. The differential diagnosis included orbital apex syndrome causedby an infectious process such as mucormycosis or a fungal species; inflammatoryprocesses, including Tolosa-Hunt syndrome; primary or metastatic malignancy;vasculitis; hypercoagulative states; and granulomatous diseases, includingsarcoidosis.
T1-weighted, fat-suppressed, contrast-enhancedcoronal magnetic resonance image demonstrating abnormal enhancement and enlargementof the cavernous sinuses bilaterally with narrowing of the cavernous segmentsof the internal carotid arteries.
T1-weighted, fat-suppressed, contrast-enhancedaxial magnetic resonance image demonstrating shaggy enhancement of the wallof the enlarged superior ophthalmic veins and filling defect in right superiorophthalmic vein, most likely representing intraluminal thrombus.
The patient was afebrile with a white blood cell count of 18 000/µL.On the day of admission, she underwent a bifrontal craniotomy and decompressionof the left optic nerve within the bony canal. Culture and biopsy specimenswere obtained from the orbits and cavernous sinus. Intraoperative findingswere remarkable for pale, firm orbital fat with multiple adhesions and considerablyswollen and tense periorbita on the left side. No frank pus was seen. Frozensection examination findings from the left orbital fat biopsy revealed anacute inflammatory infiltrate with numerous neutrophils and scattered lymphocytesand macrophages without serious vasculitis. Gram stain showed gram-positivecocci. Postoperatively, the antibiotic regimen was changed to intravenousvancomycin hydrochloride, ceftriaxone sodium, and clindamycin empirically.
Physical examination findings remained the same during the first postoperativeday. Visual acuity was counting fingers OU. Two days after surgery, the patienthad no light perception bilaterally. Fundus examination findings were normalwithout disc edema.
During the first postoperative week, she showed clinical improvementwith marked decrease in proptosis, periocular edema, and chemosis. Right opticdisc edema was noted 5 days postoperatively. Histopathological analysis ofthe surgical specimens revealed septic thrombosis in the left orbital fatand acute inflammation with numerous neutrophils and scattered lymphocytesand macrophages in dura of the cavernous sinus. The specimen from the rightorbital fat showed fat necrosis. No granulomatous, primary vasculitic, orneoplastic process was noted. Findings on chest radiography and cardiac echocardiographywere within normal limits. Results of a workup for hypercoagulability andantineutrophil cytoplasmic antibodies were negative. Culture specimens fromthe left orbit grew F nucleatum and rare αstreptococci 7 days after the operation. The F nucleatum culture results were sensitive to clindamycin. Blood culture findingsremained negative during the entire follow-up. Panorex dental radiographyrevealed localized periodontal erosive changes with loss of the bony rootof the right mandibular canine tooth. The patient was treated with intravenousclindamycin for 1 more week and was discharged after 2 weeks of hospitalization.At the time of discharge, the periocular edema, erythema, and chemosis weretotally resolved with minimal residual bilateral proptosis. Ocular motilitywas within normal limits bilaterally. Visual acuity remained no light perceptionwith nonreactive, dilated pupils bilaterally.
Fusobacterium nucleatum can form aggregateswith other bacteria in periodontal diseases and can behave synergisticallywith other bacteria in mixed infections.1 Devitalizedtissue may provide a suitable environment for the growth of these organisms.The production of proteolytic enzymes by Fusobacterium organisms may allowfor invasion of regional veins, even without tissue necrosis.2
Fusobacterium nucleatum was reported to beassociated with gingivitis, periodontal disease, abscesses, and venous thrombosisin various anatomic locations associated with septicemia.1,3 Weare unaware of previous reports of septic thrombophlebitis of the orbit andcavernous sinus caused by F nucleatum. Two hypothesescan be considered for the pathogenesis of the thrombophlebitis of the cavernoussinus: (1) recent infection of a preexisting cavernous sinus thrombosis (however,our patient's hypercoagulopathy study results were normal, and the conditionresolved with appropriate antibiotic treatment without anticoagulation); (2)more likely is that the fusobacterial infection arose in the periodontal spaceand spread secondarily to involve the cavernous sinus and orbits. This latterpossibility is supported by the fact that F nucleatum hasthrombogenic ability.4 We realize, however,that an infectious process arising primarily in the cavernous sinus cannotbe completely excluded.
Cavernous sinus thrombophlebitis can progress very rapidly—oftenin a matter of hours—and even with appropriate surgical and antibiotictreatment, it can be fatal or result in serious complications.5 Escardoet al6 reported a case of orbital cellulitiscaused by F necrophorum that required 3 urgent surgicalinterventions and 30 days of intravenous antibiotic treatment. Despite thisintensive treatment, the patient's vision did not fully recover. Early examinationof the patient with only a 1-day history of proptosis may have allowed earlydiagnosis and treatment and possibly helped to preserve sight.
This case also illustrates the prolonged period that may be requiredto isolate Fusobacterium organisms. It is therefore imperative that culturesbe allowed a protracted incubation period so that important pathogens arenot missed following isolation of rapidly growing pathogens, resulting ininappropriate antimicrobial therapy.1
Although rare, F nucleatum can be a cause ofsevere septic thrombophlebitis of the orbit and cavernous sinus, which, despiteintensive treatment, may result in severe morbidity.
The authors have no relevant financial interest in this article.
Corresponding author: Yonca Ozkan Arat Medicine, Scurlock Tower,6560 Fannin St, Suite 902, Houston, TX 77030.
Arat YO, Shetlar DJ, Rose JE. Blindness From Septic Thrombophlebitis of the Orbit and Cavernous SinusCaused by Fusobacterium nucleatum. Arch Ophthalmol. 2004;122(4):652–654. doi:10.1001/archopht.122.4.652
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