LETTERS TO THE EDITOR Striatal lesions: an underestimated complication of cerebral malaria? M. Vokaer a , J. C. Bier a , P. David b , S. Grand c , F. Supiot a , M. Ventura a,d and E. J. Bartholome´ a Departments of a Neurology and b Neuro- radiology, Erasme Hospital, Brussels, Belgium; c Department of Radiology, CHU de Grenoble, Grenoble, France; and d Center for Traumatology and Rehabilitation, Brussels, Belgium Correspondence: Dr Mathieu Vokaer, Hoˆpital Erasme, Service de Neurologie (3 e` me e´tage), 808 route de Lennik, 1070 Bruxelles, Belgium (tel.: +32 2 555 46 22; fax: +32 2 555 39 42; e-mail: mvokaer@ulb.ac.be). Keywords: CNS infection, cerebral malaria, extrapyramidal disorders, striatal necrosis Received 5 January 2004 Accepted 23 April 2004 Sirs, Cerebral malaria (CM) is characterized by altered consciousness, acute confusional state, seizures, anaemia, hypoglycaemia, brainstem involvement and retinal hae- morrhage with confirmation of Plasmo- dium parasitaemia, when other causes of encephalopathy have been excluded. (Nguyen et al., 1996; Newton and Warrell, 1998) Brain imaging in CM typically shows an increase in brain volume with or without cerebral oedema whereas focal cerebral lesions are usually not present (Newton and Warrell, 1998). We here report the first description of CM complicated by bilateral striatal lesions and propose a possible physiopathological mechanism. A 37-year-old right-handed Belgian man presented with fever (40 °C), diarrhoea, hypotension (71/43 mmHg) and anuric renal failure 5 days after his return from Kenya where he had not taken any anti- malarial prophylactic therapy. His medical history was unremarkable. Peripheral blood smear was positive for Plasmodium falciparum (40% parasitae- mia). Plasma lactate was >8 mEq/l with a pH of 7, 18. Treatment by quinine 1.5 g/day was initiated. He then presented generalized tonic-clonic seizures. Brain magnetic resonance imaging showed bilat- eral lesions affecting globus pallidus, sub- thalamic nucleus and substantia nigra (Fig. 1). When he was admitted in our rehabilitation unit 2 months later, he suf- fered from tetraparesis and parkinsonism. Neurological examination revealed sacc- adic pursuit and several extrapyramidal signs including bilateral upper limbs rigid- ity, postural tremor and bradykinesia, a tetraparesis predominantly affecting the lower limbs with bilateral lower limb hyporeflexia. There were no pyramidal or cerebellar signs. Electrophysiological investigations disclosed an ongoing axonal sensorimotor polyneuropathy suggesting intensive care unit polyneuropathy. Striatal lesions have never been des- cribed as a complication of CM. The iden- tified causes of bilateral striatal necrosis are severe metabolic acidosis, methanol intoxication, carbon monoxide intoxicat- ion, hypoxic encephalopathy, cyanide poi- soning, venous infarction, osmotic demyelination, Wilson’s disease, Leigh’s syndrome, mitochondrial encephalopathy, haemolytic-uraemic syndrome, sickle-cell disease acidosis, metformin-induced lactic acidosis, Mycoplasma pneumoniae infec- tion, Arthrinium-produced mycotoxin, glutaric acidemia and rare cases of familial striatal necrosis (Goodman and Noren- berg, 1983; He et al., 1995; Kamei et al., 1996; Mewborne et al., 1998; Faris et al., 2000; Zambrino et al., 2000). In most of these cases, the implicated pathogenic mechanism is thought to be a preferential sensibility of striatal neurons to lactic acid- osis induced by disturbance of the mitoch- ondrial metabolism (Kamei et al., 1996). Compared with these cases, our patient presented bilateral lesions of the striatum and parkinsonism almost disappeared after 1 year of follow-up. Interestingly, there are only few descriptions of extrapyramidal syndrome associated with CM (Arya et al., 1989; Chaudhary et al., 1992) or following chloroquine therapy (Singhi et al., 1979). In these cases, neither focal brain lesion were reported nor physiopathological mechan- ism proposed (Singhi et al., 1979; Arya et al., 1989; Chaudhary et al., 1992). How- ever, mild parkinsonism because of striatal lesions may have been underestimated in developing countries where CM is more frequent. In our case the co-occurrence of CM, lactic acidosis and systemic hypoten- sion may explain the severity of the clinical- radiological picture (Kamei et al., 1996). 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Serial imaging of bilateral striatal necrosis associated with acidaemia in adults. Neuroradiology 38:437–440. Mewborne JD, Ricci PE, Appel RG (1998). Cranial CT findings in metformin (Glucoph- age)-induced lactic acidosis. J Comput Assist Tomogr 22:528–529. Newton CR, Warrell DA (1998). Neurological manifestations of falciparum malaria. Ann Neurol 43:695–702. Nguyen TH, Day NP, Ly VC et al. (1996). Post-malaria neurological syndrome. Lancet 348:917–921. Figure 1 Left: magnetic resonance imaging (MRI) frontal slice showing the bilateral fluid- attenuated inversion recovery (FLAIR)-hyperintense signal in the globus pallidus. Right: MRI frontal slice showing the bilateral FLAIR-hyperintense signal in the substantia nigra. Ó 2004 EFNS 715 European Journal of Neurology 2004, 11: 715–717