Ophthalmoparesis, papillitis and premacular hemorrhage in a case with endocarditis: A rare presentation of brucellosis

We report a rare presentation of brucellosis as bilateral optic nerve and right abducent nerve involvement, and endocarditis complicated by right premacular hemorrhage in a 28-year-old white female. The patient showed improvement with both medical and surgical therapy. Brucellosis should be considered in the differential diagnosis of papillitis, gaze palsy and endocarditis complicated with premacular hemorrhage in endemic regions.

risk factor for keratitis in agricultural communities, in whom the incidence of fungal keratitis is highest in India. [6] However, all such incidents do not result in ulcer unless there is a breach in the corneal epithelium, delay in healing that provides the "window of opportunity", [7] lodgment of the microorganism and development of subsequent ulceration. Oft en it may be diffi cult to ascertain whether the sequel of corneal ulceration in this group of patients is because of direct trauma or because of the poorly healing epithelium. Clinically, it may also be diffi cult in the early stages to diff erentiate corneal erosions from infections. Further symptoms of recurrent corneal erosion in latt ice dystrophy need to be distinguished from symptoms of lid margin disease which is commonly present in the general population. Nevertheless the compromised epithelium in latt ice dystrophy is prone to frequent erosions and poor healing and thus provides the necessary milieu for the development of fungal keratitis.
In conclusion we would like to draw the att ention of all ophthalmologists to the risk of microbial keratitis in patients with latt ice and other epithelial and stromal dystrophies which compromises the corneal epithelial barrier. Patients should be advised about using protective glasses during their work to avoid entry of foreign body or mild trauma. Further, those suff ering from mild ocular surface abrasion following trauma may benefi t from prophylaxis with topical antibiotics [7] and suffi cient lubricants to promote healing.

Özlem Gürses Sahin, Aysel Pelit 1 , Tuğba Turunc 2 ,
Yonca Aydin Akova 1 We report a rare presentation of brucellosis as bilateral optic nerve and right abducent nerve involvement, and endocarditis complicated by right premacular hemorrhage in a 28-year-old white female. The patient showed improvement with both medical and surgical therapy. Brucellosis should be considered in the differential diagnosis of papillitis, gaze palsy and endocarditis complicated with premacular hemorrhage in endemic regions. Brucellosis, which is endemic in Turkey, is a systemic infection that can aff ect any organ or system in the body. [1] The most common ocular manifestations of brucellosis are considered as anterior uveitis and choroiditis. [2] We report a rare case of brucellosis with bilateral involvement of optic nerve, involvement of right abducent nerve, and endocarditis complicated by premacular hemorrhage. To our knowledge, premacular hemorrhage secondary to brucella endocarditis is the fi rst to be reported.

Case Report
A 28-year-old white female with a history of acute rheumatic fever and aortic valve insufficiency presented with poor physical condition associated with headache, fever, sweating and neck stiff ness. She also showed sudden onset of painless blurred vision in the right eye associated with diplopia. Best corrected visual acuity of the right eye was 20/400 and the left eye was 20/50. She had right aff erent pupillary defect and sluggish response to light on the left side. She had 30-45 prism diopters of right esotropia. The patient had reduced sensitivity to color vision higher in the red-green direction than in the blue-yellow direction in both eyes. The right fundus revealed 1 ½ disc diameter of premacular hemorrhage [ Fig. 1

Discussion
High index of clinical suspicion coupled with seroagglutination tests have been recommended for diagnosis of brucellosis in endemic areas. [1] The most adequate cut-off point for seroagglutination has been considered as 1/160 with a sensitivity of 93% and specifi city of 97%. [1] Seroagglutination test for brucella was positive over 1/320 dilution in our case. The incidence of neurobrucellosis has been reported to be high in endemic areas. [3] Detection of any titers of antibodies in CSF has been considered to provide evidence of neurobrucellosis. [3,4] However, CSF titer higher than 1/160 associated with increased CSF opening pressure which is greater than 200 mm H 2 O, pleocytosis, elevated protein, and reduced glucose concentrations have been considered for the diagnosis of brucella meningitis. [5] In addition, abnormal cranial CT and MRI fi ndings have been reported for cases with brucella meningitis. [5] Our case showed CSF agglutination titers for brucella over 1/32 dilution. She had an opening pressure of 150 mm H 2 O associated with normal CSF biochemistry without pleocytosis. Her cranial CT and MRI scans were normal. She had mild meningeal signs associated with right esotropia and bilateral papillitis.
She was diagnosed as having neurobrucellosis with right abducent nerve and bilateral optic nerve involvement. Cardiac involvement has been considered to be rare but potentially lethal complication of brucellosis. [6,7] Brucella endocarditis is repoted to be mostly associated with acute rheumatic fever involving the aortic valve. [6] Past medical history of our patient was signifi cant for acute rheumatic fever and aortic valve insuffi ciency. Echocardiography disclosed vegetation on the aortic valve. Premacular hemorrhage has been reported as a complication of subacute bacterial endocarditis. [7] Premacular hemorrhage detected in the right eye of our patient was considered to be related to an embolic process from the vegetation on the aortic valve. Cranial nerve involvement in neurobrucellosis has been reported previously, [8] however, neurobrucellosis and endocarditis complicated by premacular hemorrhage in our case from an endemic region was considered as a rare condition. Our patient showed dramatic clinical improvement aft er surgical removal of vegetation and replacement of aortic valve followed by specific treatment with rifampin and doxycycline for brucella. In summary, brucellosis should be considered in the diff erential diagnosis of optic neuritis, gaze palsy and endocarditis complicated by subhyaloid/preretinal hemorrhage in endemic areas.