Pure word deafness and unilateral right temporo-parietal lesions: a case report.

ing tremor was observed. Laboratory investigations were normal except for brucella agglutination titres of 1/80 and nonagglutinating antibodies titre of 1/1280 in the serum. CSF protein, glucose, cells were normal and cultures were negative. Brucella agglutination titres were only slightly positive (1/8). CT scan of the head disclosed no abnormality. Brucella melitensis was isolated from two blood cultures performed when the patient had no fever. After levodopa and bromocriptine withdrawal, treatment with doxycycline by mouth 100mg bid, streptomycin 1 g IM daily and rifampin 600mg daily was introduced. One month later, the patient was able to walk without assistance, but still had a mild hypokinesia and resting tremor. Doxycycline and rifampin were continued for 2 months, and the patient was discharged free of symptoms. According to Roger and Poursine's classification, our patient suffered a late onset neurobrucellosis presenting as Parkinsonism. Though considered rare, extrapyramidal forms of neurobrucellosis have been well recorded, especially in Italian publications4; amongst these were two cases of subacute hemiparkinsonism.4 I Unlike ours, both may be included in the early form of Roger and Poursine's, their symptoms having appeared during the course of brucellosis. In both cases, right hemiparkinsonism persisted because treatment was limited by streptomycin toxicity and the lack ofnew antibiotics at the time. The exact pathogenisis of Parkinsonism in brucellosis is still unknown; there is a lack of pathological studies. Gianelli4 suggest a focal encephalitis with circumscribed symptoms, related to concurrent treatment (there being normal CSF), probably due to an idiosyncratic immunological reaction. In our opinion, Parkinsonism unresponsive to levodopa and bromocriptine suggests a post-synaptic dopamine receptor lesion, such as corpus striatum vascular inflammation similar to that observed in syphilitic Parkinsonism.6 Whether this reaction is immunological and not due to direct invasion by the organism, cannot be determined only on the basis of a negative CSF culture and agglutination titre, because serine pre-treatment CSF cultures obviously are not performed. Besides, CSF agglutination titres in chronic cases are usually low, and non-agglutinating antibodies or complement-fixation titres need to be included in adequate serologic study.2 Finally, we believe that previous treatment is the reason for the lack of CSF abnormality, as found in modern or partially treated neurosyphilis,7 especially in chronic and encephalitic forms. All these considerations lead us to recommend serine blood cultures when suspecting brucellosis in patients with unusual neurological manifestations and normal or inconvincing CSF changes, even if fever or other general symptoms are absent. Treatment with two, three or even four drugs, including those with high CSF penetration (rifampin, trimethoprimsulfamethoxazole), seems to be desirable8 but there are neither comparative studies nor large series to verify the best drug combination. We conclude by emphasising the importance of this sometimes underestimated disease, with a marked CNS affinity, which is still far from being eradicated.