Brain Advance Access originally published online on February 15, 2007
Brain 2007 130(3):816-827; doi:10.1093/brain/awl347
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Atypical parkinsonism in Guadeloupe: a common risk factor for two closely related phenotypes?
Departments of 1Neurology and 2Radiology, University Hospital of Pointe-à-Pitre, Guadeloupe, F.W.I., 3INSERM, UMR679, Experimental Neurology and Therapeutics, Hôpital de la Salpêtrière, Université Pierre et Marie Curie-Paris 6, 4Federation of Neurology, Hôpital de la Salpêtrière, Paris, France and 5Department of Neurology, Philipps University, Marburg, Germany
Corresponding author: Dr Annie Lannuzel, Department of Neurology, University Hospital of Pointe-à-Pitre, BP465, 97 159 Pointe-à-Pitre Cedex, Guadeloupe, F.W.I. E-mail: annie.lannuzel{at}wanadoo.fr
In Guadeloupe, there is an abnormally high frequency of atypical parkinsonism. Only one-third of the patients that develop parkinsonian symptoms were reported to present the classical features of idiopathic Parkinson disease and one-third a syndrome resembling progressive supranuclear palsy (PSP). The others were unclassifiable, according to established criteria. We carried out a cross-sectional study of 160 parkinsonian patients to: (i) define more precisely the clinical phenotypes of the PSP-like syndrome and the parkinsonism that was considered unclassifiable in comparison with previously known disorders; (ii) define the neuropsychological and brain imaging features of these patients; (iii) evaluate to what extent a candidate aetiological factor, the mitochondrial complex I inhibitor annonacin contained in the fruit and leaves of the tropical plant Annona muricata (soursop) plays a role in the neurological syndrome. Neuropsychological tests and MRI were used to classify the patients into those with Parkinson's disease (31%), Guadeloupean PSP-like syndrome (32%), Guadeloupean parkinsonism–dementia complex (PDC, 31%) and other parkinsonism-related disorders (6%). Patients with a PSP-like syndrome developed levodopa-resistant parkinsonism, associated with early postural instability and supranuclear oculomotor dysfunction. They differed, however, from classical PSP patients by the frequency of tremor (>50%), dysautonomia (50%) and the occurrence of hallucinations (59%). PDC patients had levodopa-resistant parkinsonism associated with frontosubcortical dementia, 52% of these patients had hallucinations, but, importantly, none had oculomotor dysfunction. The pattern of neuropsychological deficits was similar in both subgroups. Cerebral atrophy was seen in the majority of the PSP-like and PDC patients, with enlargement of the third ventricle and marked T2-hypointensity in the basal ganglia, particularly the substantia nigra. Consumption of soursop was significantly greater in both PSP-like and PDC patients than in controls and Parkinson's disease patients. In conclusion, atypical Guadeloupean parkinsonism comprises two forms of parkinsonism and dementia that differ clinically by the presence of oculomotor signs, but have similar cognitive profiles and neuroimaging features, suggesting that they may constitute a single disease entity, and both were similarly exposed to annonaceous neurotoxins, notably annonacin.
Key Words: Annonaceae; annonacin; atypical parkinsonism; dementia; MRI; PSP
Abbreviations: DLB, dementia with Lewy bodies; FAB, frontal assessment battery; FTDP, frontotemporal dementia with parkinsonism; Gd-PDC, guadeloupean parkinsonism-dementia-complex; Gd-PSP, guadeloupean progressive supranuclear palsy; I-PD, idiopathic Parkinson disease; Mattis DRS, Mattis Dementia Rating Scale; MMSE, Mini-Mental Status Examination; PSP, progressive supranuclear palsy; PDC, parkinsonism-dementia complex; UPDRS, Unified Parkinson's Disease Rating Scale
Received July 25, 2006. Revised October 19, 2006. Accepted November 15, 2006.
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