Discussion
Brucella species are intracellular pathogens causing systemic infection in humans and livestock. Brucellosis can involve the central nervous system through direct damage or indirect induction of immune neuroinflammation (7). Neurobrucellosis usually presents with classic meningoencephalitis syndrome. However, we should consider the diagnosis in any patient presenting with chronic headache or neuropsychiatric symptoms, even without signs of meningeal irritation or fever (8). Neurobrucellosis is also suspected in patients with chronic neurological symptoms accompanied by CSF lymphocytosis or compatible neuroimaging findings. Nevertheless, it is confirmed by positive serum brucella agglutination test, positive serological tests (increased brucella antibody in the CSF), positive CSF Wright test, and isolation of Brucella species or detection of brucella DNA in the CSF with polymerase chain reaction (PCR) test (9). Our patient had been previously diagnosed with idiopathic PTC. Nonetheless, her aggravating symptoms and the addition of systemic manifestations like fever and myalgia suspected us of the diagnosis of brucellosis. In addition, the CSF lymphocytosis made our suspicion to neurobrucellosis stronger.
PTC has been a rare presentation of brucellosis. PTC is characterized by elevated ICP with normal CSF cell count, biochemistries, and normal brain imaging findings. It has been more commonly reported in females of childbearing age and obese individuals(10, 11). PTC may be primary or secondary to traumatic brain injuries, collagen vascular disorders like systemic lupus erythematosus (SLE), medications like tetracyclines, isotretinoin, phenytoin, and other medical conditions (12-15). Our patient was initially diagnosed as being involved with primary or idiopathic PTC. However, brucellosis was later identified as the causative condition for her problem. Previously, rarely reported infections as the underlying causes of PTC included measles, varicella, Lyme disease, human immunodeficiency virus (HIV), and tuberculosis (16-21). However, brucellosis has been more commonly reported recently as the trigger for PTC, compared with other mentioned infections (6, 22-26). As aforementioned, neuroimaging usually offers normal findings in PTC.
Nonetheless, flattening of the globe, empty sella, enlarged prelaminar optic nerve or optic nerve sheath (ONS), enhanced optic nerve head, increased tortuosity of the optic nerve, distension of the perioptic subarachnoid space, narrowing of Meckel’s cave and sinovenous stenosis may be identified as indicators of PTC (27-29). Treatment of PTC is based on controlling the underlying condition, medical management, repeated lumbar punctures, and surgical interventions(11). However, PTC in the settings of brucellosis can readily be controlled by antibiotic therapy with favorable results if timely diagnosed and not led to sequels.
On the other hand, delayed diagnosis and management of PTC may result in poor visual outcomes (30). The best therapeutic regimen has not been determined for neurobrucellosis. Nevertheless, at least a dual- or triple therapy with a combination of streptomycin, ceftriaxone, doxycycline, rifampicin, and trimethoprim-sulfamethoxazole for at least 6 months is recommended. Ceftriaxone-based therapeutic regimens have yielded the highest cure rates for neurobrucellosis(31). Our patient was initially responsive to CSF volume-decreasing pharmaceuticals but was later found to require surgical management. Nevertheless, she was promptly started on combination antimicrobial therapy after brucellosis was recognized as the underlying cause of her condition. Fortunately, she responded favorably to medical treatment and was deprived of the need to undergo surgical shunt implantation.