Rev Bras Neurol. 53(1):5-14, 2017 NEUROPSYCHIATRIC MANIFESTATIONS AND EPIDEMIOLOGY OF NEUROCYSTICERCOSIS

MANIFESTAÇÕES NEUROPSIQUIÁTRICAS E EPIDEMIOLOGIA DE NEUROCISTICERCOSE

Ericson Dametto1

ABSTRACT RESUMO Neurocysticercosis (NCC) is the brain caused by larval stages Neurocisticercose é a infecção cerebral causada pelos estágios lar- of the helminth . vais do helminto Taenia solium. The embryos of Taenia travel through the bloodstream and can reach Os embriões da Taenia deslocam-se através da corrente sanguínea e the brain, muscles, eyes, and various organs. In the brain, the psy- podem atingir o cérebro, músculos, olhos e vários órgãos. No cére- chiatric manifestations are mood disorders, depression and anxiety, bro, as manifestações psiquiátricas são transtornos de humor, de- which are commonly associated with and sensory-motor pressão e ansiedade, as quais estão comumente associados com GHȴFLWV HSLOHSVLDHGHȴFL¬QFLDVVHQVµULRPRWRUDV Neurocysticercosis is a frequent parasitic disease in the world popu- Neurocisticercose é uma parasitose frequente na população mun- lation; it is endemic in Central and South America, Asia and Sub-Saha- GLDO«HQG¬PLFDQD$P«ULFD&HQWUDOHGR6XOƒVLDHƒIULFDVXEVDDULD- ran Africa. In the present review, we report the major symptoms and na. Na presente revisão, relatamos os principais sintomas e sinais de signals of neurocysticercosis common to neurological and psychiatric neurocisticercose pertinentes a doenças neurológicas e psiquiátricas. LOOQHVVHV :H EULHȵ\ SUHVHQW (SLGHPLRORJ\ RI WKRVH PDQLIHVWDWLRQV 1µV EUHYHPHQWH DSUHVHQWDPRV D (SLGHPLRORJLD GHVVDV PDQLIHVWD- and analyze the relationship between pathological changes and NCC ções, e analisamos a relação entre alterações patológicas e sintoma- symptomatology. tologia da NCC. Objectives and Methodology. A literature review was conducted to Objetivos e Metodologia. Uma revisão da literatura foi conduzida characterize epidemiological, neurological and psychiatric manifesta- para caracterizar a epidemiologia, as manifestações neurológicas e WLRQVRI1&&7KHȴQDOSDSHUVZHUHVHOHFWHGRIDVHWRISXEOL- SVLTXL£WULFDVGH1&&2VDUWLJRVȴQDLVIRUDPVHOHFLRQDGRVGHXP FDWLRQVIURPWR FRQMXQWRGHSXEOLFD©·HVHQWUHD Results. NCC is a major cause of epilepsy in endemic areas; further- Resultados. NCC é uma importante etiologia de epilepsia em áreas PRUHOHDGVWRDGLYHUVLW\RIPRWRUDQGVHQVLWLYHGHȴFLWVPDQLIHVWD- HQG¬PLFDVDO«PGLVVRFDXVDXPDGLYHUVLGDGHGHGHȴFL¬QFLDVPRWR- tions vary from headache to severe intracranial hypertension. ras e sensoriais, as manifestações variam de cefaleia a severa hiper- Potentially fatal conditions include arteritis, encephalitis and hydro- tensão intracraniana. cephalus. Condições potencialmente fatais incluem arterites, encefalites e hi- Depression and cognitive decline remain among the most important drocefalia. psychiatric manifestations. Depressão e declíneo cognitive permanecem entre as mais importan- 1HXURSV\FKLDWULF PDQLIHVWDWLRQV (SLGHPLRORJ\ DQG QHXURLPDJLQJ tes manifestações psiquiátricas. provide diagnostic criteria. Brain scans may reveal one or diverse cys- Manifestações neuropsiquiátricas, epidemiologia e neuroimagem WVȴOOHGZLWKȵXLGZLWKLQDVFROH[ SDUDVLWHȇVKHDG  SURY¬P RV FULW«ULRV GH GLDJQµVWLFR $V LPDJHQV FHUHEUDLV SRGHP Conclusion.1&&ȇVGLYHUVLW\RISUHVHQWDWLRQVHQFRXUDJHKHDOWKSUR- revelar um ou diversos cistos preenchidos com líquido e o escólex fessionals to consider it in diagnoses, especially in endemic countries, (cabeça) do parasito. and also in non-endemic areas because migrants and travelers are Conclusões. A diversidade de apresentações da NCC encoraja os subject to contagious. SURȴVVLRQDLVGHVD¼GHDFRQVLGHU£ODGHQWUHRVGLDJQµVWLFRVHVSH- Treatment consists in use of (albendazol, prazi- FLDOPHQWHHPSD¯VHVHQG¬PLFRVHWDPE«PHP£UHDVQ¥RHQG¬PLFDV quantel) and drugs to treat associated conditions (, pois migrantes e viajantes estão sujeitos ao contágio. ). Surgery is reserved to extirpate the parasite from O tratamento consiste no uso de antiparasíticos (albendazol, prazi- SDUWLFXODUORFDWLRQV H\HVVSLQDOFRUGFHUHEUDOYHQWULFOHV RUWRGL΍H- quantel) e medicamentos para tratar condições associadas (anticon- rentiate NCC from tumors, tuberculosis, mycosis, etc. vulsivantes, corticosteróides). Cirurgia é reservada para remoção do Prevention includes treatment of intestinal , sanitation parasito de locais particulares (olhos, medula espinhal, ventrículos in animal farming, food preparing hygiene, quality control of water cerebrais) ou para diferenciar NCC de tumores, tuberculose, micose, and food. etc. Prevenção inclui o tratamento de helmintíases intestinais, sanidade Key words: Neurocysticercosis, epilepsy, cognitive decline, depres- animal, higiene ao preparar alimentos, controle da qualidade da água sion, encephalitis and . e alimentos.

Palavras-chaves: Neurocisticercoses, epilepsia, declínio cognitivo, depressão, encefalite e hidrocéfalo.

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Revista Brasileira de Neurologia » Volume 53 » Nº 1 » Jan/Fev/Mar 2017 5 Dametto E

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6 Revista Brasileira de Neurologia » Volume 53 » Nº 1 » Jan/Fev/Mar 2017 Neurocysticercosis neuropsychiatry

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Revista Brasileira de Neurologia » Volume 53 » Nº 1 » Jan/Fev/Mar 2017 7 Dametto E

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8 Revista Brasileira de Neurologia » Volume 53 » Nº 1 » Jan/Fev/Mar 2017 Neurocysticercosis neuropsychiatry

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10 Revista Brasileira de Neurologia » Volume 53 » Nº 1 » Jan/Fev/Mar 2017 Neurocysticercosis neuropsychiatry

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Table 1. Epidemiology of NCC

Locations Authors

The frequency of NNC ranged from 0.2% to 52% worldwide, and its association with epilepsy ranged from 0.11% to [53] 1.32%.

Worldwide World Health Organization estimates 50 million cases worldwide, it causes about 50,000 deaths each year. [54]

NCC causes approximately 5 million cases of epilepsy in the world. [55]

Latin America It was estimated infection in about 350,000 individuals, in Latin America. [56]

NCC was the cause of epilepsy in up to 50% of Indian patients presenting with partial . It was also a major cause of Asia [57] epilepsy in Bali (Indonesia), Vietnam and possibly China and Nepal.

West Africa In Togo and Benin, the prevalence of cysticercosis was 2.4% and 1.3%, respectively. [58]

Human cysticercosis was characterized as endemic in Rwanda, Burundi, the Democratic Republic of Congo and Cameroon. [58] Cysticercosis shown to be one of the major causes of epilepsy in Cameroon with figures as high as 44.6%. Central Africa Cysticercosis was present in 7% of 300 autopsies carried out in a region of Butare. [58]

Cases of NCC was 176 in 17 European countries (Austria, Belgium, Denmark, Finland, France, Germany, Greece, Hungary, Europe Ireland, Italy, Latvia, Netherlands, Sweden, United Kingdom, and Croatia, Norway, Switzerland). A particular epidemic [59] situation was described in Spain and Portugal.

NC has emerged as a serious public health problem. [60]

United States of America Data in the Nationwide Inpatient Sample for 2003–2012 estimated 18,584 hospitalizations for NCC (charges >US $908 million). The hospitalization was highest among Hispanics. The charges to cysticercosis exceeded those for malaria and [61] were greater than for other neglected tropical diseases combined.

Canada Literature reported 60 cases, in the past two decades. [62]

Cysticercosis was present in 2.4% of autopsies in Mexico (n. 20,026). [63] Mexico Serological studies revealed infection rate from 4.9 to 12.2% for human cysticercosis, in rural areas, the prevalence was [64, 65] 9.1% as determined by CT.

Prevalence of NCC was approximately 24% in individuals hospitalized for diagnosis of epilepsy in Chapecó/SC [66]

In Uberaba-MG, cysticercosis frequency in autopsies was 3.3%, the brain location was 79.2% (n. 53, age 15-86 years). [67] Brazil Percentage of NCC was 5.1% in patients with epileptic seizures, in Recife City. [68]

Brazilian literature showed incidence of 1.5% in autopsies and 3.0% in clinical trials. [69]

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Table 2. Psychiatric manifestations of NCC

Manifestations Authors

Percentage of depression was higher than in the general population, as follows: NCC with epilepsy (83%), [12] NCC without epilepsy (88%), sample size 65 patients. Depression Depression was the most frequent psychiatric diagnosis (52.6%), in a sample of 38 patients. [13]

These diseases were the most common in 50 patients with NCC and epilepsy, compared to 50 patients with Mixed anxiety and depression epilepsy only. Psychiatric disorders had frequency of 68% in patients with NCC and epilepsy, compared to [70] 44% of those only with epilepsy. Left sided lesions had greater psychiatric morbidity.

Decline in cognitive function Decline in cognitive function was present in older children with NCC, sample size 83 patients. [71]

Dementia was found in 1.3% of NCC patients, sample size 592. [72]

Manic syndrome Case report of manic syndrome secondary to NCC was responsive to risperidone. [73]

Psychotic symptoms Case report of NCC presenting as delusion. [74]

Schizophrenia Case report of NCC presenting as schizophrenia. [75]

Case report with negative self-evaluation, low self-stem, feelings of shame directed to the diagnosis of the Personality changes [76] NCC.

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Table 3. Neurologic manifestations of NCC

Manifestations Authors

Seizures were generalized in 121 patients and partial in 82. CT showed parenchymal brain calcifications in 53 Seizure and epilepsy [77] patients and cysts in 150. Use of anticysticercal drugs improved seizure control.

Headaches occur: Headache a. as migraine and tension-type, [78] b. as result of increased intracranial pressure.

Focal deficits were the third most frequent manifestation, behind seizures or epilepsy and headache, in a Sensory-motor Deficits [20] systematic review.

The majority of patients presented with a chronic and relatively normotensive hydrocephalus, in a sample of 11 Hidrocephalus patients. Impairment of CSF flow required permanent CSF shunting. Exceptionally, one cyst was removed by [79] surgery.

It was reported three cases of stroke secondary to neurocysticercosis. MRI demonstrated cortical and subcortical Cerebrovascular disease infarction areas and cisternal cysts. Angiographic showed arteritis of basilar and carotid arterial system. [80] Infarts happened in small arteries in most cases, but middle cerebral and carothid arteries can be affected. In the spinal regions cervical and lumbosacral was found cystic lesions, in a patient. and anti- inflammatory treatment was initiated with albendazol (2×400 mg/day) and steroids (prednisone 60 mg/day) for 4 [81] weeks. The patient was retreated. Spinal cysticercosis presented mainly with motor symptoms (21/27 patients): paraparesis and paraplegia were the Spinal cord lesions [82] most common signs; one-third of patients had sphincter dysfunction.

Intramedullary spinal cord neurocysticercosis presenting as Brown-Séquard syndrome, i.e. , paralysis and loss of [83] proprioception on the same side as the lesion, and loss of pain and temperature sensation on the contralateral side.

The signs of cysticercosis in the sellar region include headache, vision loss, hypopituitarism, seizures, and Neuroendocrine syndromes [84] meningitis.

Papilledema Case report of papilledema due to NCC in brain ventricle. [85]

Case report due to NCC lesion in the midbrain characterized by contralateral hemiataxia and oculomotor cranial Claude’s syndrome [86] nerve palsy.

The main histological alterations in neucorysticercosis are edema, perivascular infiltrate, gliosis, fibrosis, Chronic inflamation in the brain tissue granulomatosis and calcification. Chronic inflammatory reaction are responsible for NCC alterations in the human [87] brain.

Case report of non-communicating hydrocephalus with headaches, ataxia and loss of vision due to intraventricular Loss of vision and ataxia [88] cyst.

Dizziness and ataxic gate Case report of a rare racemose cysticercose in the cerebellar hemisphere. [89]

A patient with parkinsonism secondary to NCC. Scans showed edema in the midbrain. Parkinsonism symptoms Parkinsonism were exacerbated after albendazole treatment. Symptoms improved after methylprednisolone pulse therapy for 5 [90] days, and levodopa/carbidopa for eight months.

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