Introduction: Toxocariasis is a worldwide human helminthiasis, which is mostly asymptomatic and caused by toxocara canis, a roundworm in dogs. These can cause visceral larva migrans syndrome in humans who ingest contaminated soil. CNS manifestation with a focal mass lesion is very rare, seizures often being the first symptom. Case report: We describe an 11-year-old girl presenting with a generalized epileptic seizure and eosinophilia in blood. Under antibiotic therapy under the assumption of toxoplasmosis the lesion did not decrease and surgical resection was considered. We used computer-assisted surgery (CAS) for careful tissue resection. Postoperatively the diagnosis of toxocariasis was confirmed and albendozole medication was administered for 7days. The patient developed well without neurological deficits or seizures. Conclusion: We conclude that although neurological involvement is rare in toxocariasis, a cerebral infection in a child with epileptic seizures and eosinophilia should be considere

Bächli, Heidi

Gratzl, Otmar

Minet, Jean

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Childs Nerv Syst (2004) 20:468–472DOI 10.1007/s00381-004-0963-x O R I G I NA L PA P ERHeidi BchliJean Claude MinetOtmar GratzlCerebral toxocariasis: a possible causeof epileptic seizure in childrenReceived: 9 January 2004Published online: 12 May 2004 Springer-Verlag 2004H. Bchli ()) · O. GratzlDepartment of Neurosurgery,University Hospital Basel,Spitalstrasse 21, 4031 Basel, Switzerlande-mail: hbaechli@uhbs.chFax: +41-61-2657138J. C. MinetDepartment of Pediatrics,University Children’s Hospital Basel,Basel, SwitzerlandAbstract Introduction: Toxocariasisis a worldwide human helminthiasis,which is mostly asymptomatic andcaused by toxocara canis, a round-worm in dogs. These can cause vis-ceral larva migrans syndrome inhumans who ingest contaminatedsoil. CNS manifestation with a focalmass lesion is very rare, seizuresoften being the first symptom.Case report: We describe an 11-year-old girl presenting with a generalizedepileptic seizure and eosinophilia inblood. Under antibiotic therapy underthe assumption of toxoplasmosis thelesion did not decrease and surgicalresection was considered. We usedcomputer-assisted surgery (CAS) forcareful tissue resection. Postopera-tively the diagnosis of toxocariasiswas confirmed and albendozolemedication was administered for7 days. The patient developed wellwithout neurological deficits or sei-zures. Conclusion: We conclude thatalthough neurological involvement israre in toxocariasis, a cerebral infec-tion in a child with epileptic seizuresand eosinophilia should be consid-ered.Keywords Cerebral toxocariasis ·Epileptic seizure · Brain abscess ·Visceral larva migrans (VLM) ·Computer-assisted surgery (CAS)IntroductionToxocariasis is a parasitic infection mainly caused byToxocara canis, an intestinal nematode (roundworm) indogs and cats, which are routinely infected. The diagnosisis based on serological findings. Toxocariasis is aworldwide health problem. The highest seroprevalencewas reported in a village of Santa Lucia (West Indies),with 86% infected children aged 6 months to 6 years.Seropositive rates differ in various countries and werehigher in tropical regions. Laufer [7] reported rates, e.g.,in the Netherlands of 19%, Germany 2.5%, Brazil 39%,Cuba 5.2%, Columbia 47.5%, and Nepal 81%. The higherthe rate of infected dogs and the easier their access topublic places, the easier it is for humans to ingest theinfected eggs. Young children are especially at high riskbecause of their play habits and tendency to put theirfingers in their mouths. Mortality in toxocariasis is un-usual, but cases of sudden allergic asthma were suggested.There is no racial predilection. Boys usually have a higherprevalence than girls, perhaps because of different playbehaviour.Toxocariasis is an aberrant infection for humans be-cause humans are only incidently hosts on whom theparasites cannot completely mature. Most cases of toxo-cariasis are asymptomatic. There are three clinical forms,including visceral larva migrans (VLM; general, derma-tologic, pulmonary, hepatic and lymphatic, rheumato-logic, cardiac, and central nervous system), ocular larvamigrans (OLM), and the covert form (less specific syn-drome).The infection of the central nervous system can causeeosinophilic meningitis [4], encephalitis or meningo-en-cephalitis [13], encephalitis with vasculitis [2, 10, 14],arachnoiditis, and spinal cord lesions [3, 6, 12, 15]; cog-nitive disorders have also been described [11].The tissue damage is due to the host inflammatoryreaction rather than the infection itself [7]. The larvaeproduce glycosylated proteins, which induce a Th2-typeCD4+ cellular immune response with production of im-469munoglobulin E (IgE) and interleukin 5. These promoteeosinophil differentiation and vascular adhesion. Migra-tion of the larva to the brain parenchyma with solitarymass lesions is very rare and there are only a few reports[5, 8, 9, 13, 16, 17, 19, 20]. Treatment options are con-troversial in the literature. We report here a case of cere-bral toxocariasis, which caused epileptic seizures, and thetherapeutic options are discussed.Case reportWe report a case of an 11-year-old girl with her first generalizedepileptic seizure. The CT scan showed a hypodense cystic lesion,right parietal, with a diameter of 2 cm with perifocal edema. Noother lesions were found on MRI (Fig. 1a), abdominal ultrasound,or chest radiography. Echocardiography showed normal myocardialfunction and on the EEG slow theta and delta waves were seen rightparieto-occipital. Biochemical and hematological tests were normalexcept for eosinophilia of 9.5%. Cerebrospinal fluid examinationwas normal without pleocytosis. A further search for parasites incerebrospinal fluid and feces was negative. Serum titers of toxo-plasmosis, cryptococcus neoformans, and echinococcus were alsonegative. Only the ELISA test for toxocara canis was positive(result received postoperatively). The patient was treated withtrileptal (2150 mg/day). Under the assumption of a brain abscesswith toxoplasma gondii, she received additional antibiotics (cef-triaxon and metronidazolum) for 42 days. The follow-up MRI(Fig. 1b) showed an increasing edema and persistence of the lesion.Therefore, we decided to remove this lesion. Computer-assistedsurgery (CAS) based on CT images was used for preoperativeplanning of the surgical approach as well as for augmented intra-operative orientation and targeting (SPOCS, Aesculap AG, Tut-tlingen, Germany) described in Fig. 2. Skin markers were applied toobtain CT image registration shown in Fig. 3. Registration accuracyhas been determined within a range of 1–2 mm and was found to besufficient for targeting small lesions [18]. The lesion was com-pletely removed. The histopathological result showed a granulo-matous inflammatory process containing large numbers of eosino-phils and neutrophils caused by a parasite. Toxocariasis was ret-rospectively suggested because of the seropositive Elisa IgG forToxocara canis. Remnants of dead parasites were not found. Thepatient received albendozole 400 mg/day for 7 days. The postop-erative MRI 1 year later (Fig. 1c) showed no pathological en-Fig. 1a–c Toxocariasis lesion right parietal in an 11-year-old girl.a Axial postcontrast T1-weighted MRI scan showing a thin ringenhancement right parietally without edema. b Axial MRI scan6 weeks later with a thick diffuse ring enhancement and perifocaledema. c Postoperative axial MRI scan (1 year later) showing nopathological gadolinium enhancementFig. 2 Computer-assisted surgery (CAS) system (Aesculap AG,Tuttlingen, Germany). On the left side is the infrared camera (Po-laris, Northern Digital, Ontario, Canada) and on the right theworkstation (Dell) with coronal, sagittal, and axial CT scans, and athree-dimensional view of the head with skin markers470hancement. The follow-up of the patient was uneventful, there wereno more epileptic seizures so the antiepileptic medication wasstopped 6 months later.Afterwards, the parents of the girl reported that friends visitedthem some weeks before hospitalization, with a dog that had notbeen sufficiently vaccinated against parasites.DiscussionCerebral toxocariasis is mainly described in the litera-ture in the form of eosinophilic pleocytosis with menin-gitis, encephalitis, or myelitis and arachnoiditis. Patho-logical CT or MRI with solitary mass lesions were onlydescribed in seven cases [5, 8, 9, 13, 16, 17, 19] andare summarized in Table 1. Seizures were often the firstFig. 3 Intraoperative view withthree dimensional navigation ofthe lesion during surgeryTable 1 Literature review of cases with cerebral toxocariasis and pathological CT/MRI findingsCase Reference Age (years) Sex Symptoms Imaging Therapy1 [9] 41 Boy Acute hemiparesis, rightepileptic fits, aphasiaLeft frontal mass lesion Surgery + albendozole2 [8] Accidental, fire victim Autopsy3 [13] Focal epileptic fit CT normal, MRI mass lesion4 [19] 54 Girl MRI several subcortical andwhite matter lesions in bothlobes5 [5] 2.5 Death from non-accidentalinjuryAutopsy6 [17] 74 Girl Focal epileptic fit Granulomatous lesions7 [16] 2 Boy Headache, mental confusion,progressive weaknessSubcortical lesion right frontal Thiabendazole ! ineffec-tive; albendozole and cor-ticosteroids471References1. Arpino C, Gattinara GC, Piergili D,Curato P (1990) Toxocara infection andepilepsy in children: a case-controlstudy. Epilepsia 31:33–362. Dousset V, Sibon I, Menegon P (2003)Case no 6. Cerebral vasculitis due totoxocara canis (or catis origin). J Radiol84:89–913. Goffette S, Jeanjean A, Duprez T,Bigaignon G, Sindic C (2000) Eosino-philic pleocytosis and myelitis relatedto toxocara canis infection. Eur JNeurol 7:703–7064. Gould M, Newell S, Green SH, GorgeRH (1985) Toxocariasis and eosino-philic meningitis. Br Med J 291:1239–12405. Hill IR, Denham DA, Scholtz CL(1985) Toxocara canis larvae in thebrain of a British child. Trans R SocTrop Med Hyg 79:351–3546. Kumar J, Kimm J (1994) MR in Tox-ocara canis myelopathy. Am J Neuro-radiol 15:1918–19207. Laufer M (2002) Toxocariasis. http://www.emedicine.com/ped/top-ic2270.htm, February 1, 20028. Nelson I, Frost JL, Schochet SS Jr(1990) Unsuspected cerebral Toxocarainfection in a fire victim. Clin Neu-ropathol 9:106–1089. Oktar N, Barcin E, Kazandi AC,Korkmaz M (2002) Cerebral Toxocaramimicking a malignant glioma. NorolBil D 19:2. http://www.med.egeedu.tr/norolbil/2002/NBD19202.htm10. Oujamaa L, Sibon I, Vital A, MenegonP (2003) Cerebral vasculitis secondaryto toxocara canis and fasciola hepaticaco-infestation. Rev Neurol (Paris)159:447–45011. Richartz E, Buchkremer G (2002) Ce-rebral toxocariasis: a rare cause ofcognitive disorders. A contribution todifferential dementia diagnosis.Nervenarzt 73:458–46212. Russegger L, Schmutzhard E (1989)Spinal toxocaral abscess. Lancet 12:39813. Ruttinger P, Hadidi H (1991) MRI incerebral toxocaral disease. J NeurolNeurosurg Psychiatry 54:361–36214. Sommer C, Ringelstein EB, Biniek R,Glockner WM (1994) Adult Toxocaracanis encephalitis. J Neurol NeurosurgPsychiatry 57:229–231symptom. In 1990, Arpino [1] showed in a case-controlstudy a significant association between seropositivity foranti-toxocara canis and seizures. The closest correlationwas seen in children less than 5 years of age and there wasa highly significant association between dog ownershipand toxocariasis. As mentioned before, toxocariasis is apublic health problem and the prevalence of infection indifferent communities is directly proportional to the in-fection rates among canines and the free access of dogs topublic places [7]. The real prevalence of toxocariasis isdifficult to estimate because most infections are asymp-tomatic and tests are only performed when diagnosis issuspected. Although people of all ages are at risk, themore severe symptoms occur mainly in young childrenaged between 1 and 7 years [7]. Dogs are one of the mainhosts. As in our case, an insufficiently vaccinated dogbelonging to friends may transfer this infection. Althoughcerebral toxocariasis is a very rare disease we must con-sider the possibility of such an infection when the childsuddenly develops epileptic seizures in combination witha contrast-enhancing lesion on a CT scan or MRI. In theliterature there are no unique therapeutic options, espe-cially surgical ones, which makes a decision in individualcases difficult. Our patient showed a persistence of thelesion; therefore, the decision to neurosurgically removeit to reveal the diagnosis was clear. We believe that atleast in cases of suspected cerebral toxocariasis a biopsyis justified to reach the diagnosis. We used CAS forcareful removal.Most patients with toxocariasis recover without ther-apy. Medical treatment with anthelmintic agents is indi-cated for severe complications in, for instance, the brain,lung, and heart [7]. Treatment of covert toxocariasisshould be individualized. We must be aware of the sideaffects of anthelmintic therapy because of a possible in-flammatory reaction and progression of edema due toHerxheimer’s reaction, so that the use of corticosteroids issometimes necessary from the beginning of the therapy.No studies have confirmed that combined treatment issuperior to single therapy [3].The anthelmintic agents that are used most are: al-bendozole (Albenza), mebendazole (Vermox), thiaben-zole (Mintezol) and diethylcarbamazine (Hetrazan).Although a brain abscess with toxocariasis is a veryrare disease, the toxocariasis itself is a widespread in-fection. The prevalence of human toxocariasis is a re-flection of the rates of infestation of dogs and hygiene inpublic places. Prevention is a public health issue. In de-veloped countries it should be obligatory for veterinariansto examine pets and deworm them. Dogs should be keptaway from playgrounds, so that the contact of childrenwith animal excrements can be minimized. Also eatingwith dirty hands, and the consumption of raw vegetablesor undercooked giblets should be avoided if possible. Inunderdeveloped countries these recommendations may bedifficult to realize. However, the knowledge of how theyinfect humans, together with sanitary education, may behelpful for the prevention of this infection.ConclusionWe conclude that although neurological involvement israre in toxocariasis, a cerebral infection should kept inmind in a child with epileptic seizures and eosinophilia.The high prevalence of Toxocara canis infestation in dogsresulting in ground contamination and the predisposi-tioning of children to infection is a public health problem.Prevention and patient education is the basis for thecontrol of this disease. Surgical resection, especially fordiagnosis, should be considered.47215. Strupp M, Pfister HW, Eichenlaub S,Arbusow V (1999) Meningomyelitis ina case of toxocariasis with markedlyisolated CSF eosinophilia and an MRI-documented related thoracic cord le-sion. J Neurol 246:741–74416. Vidal JE, Sztajnbok J, Seguro AC(2003) Eosinophilic meningoencephali-tis due to Toxocara canis: case reportand review of the literature. Am J TropMed Hyg 69:341–34317. Viovy A, Jofr L, Noemi I, Erazo R,Cerva J (1999) Granuloma cerebral portoxocara: comunicacion de un casoclinico. Rev Chil Infectol 15:312–31618. Westermann B, Hauser H (2000) Onlinehead motion tracking applied to thepatient registration problem. ComputAided Surg 5:137–14719. Xinou E, Lefkopoulos A, Gelagoti M,Drevelegas A, Diakou A, Milon Dimi-triadis AS (2003) CT and MRI imagingfindings in cerebral toxocaral disease.Am J Neuroradiol 24:714–71820. Zachariah SB, Zachariah B, Varghese R(1994) Neuroimaging studies of cere-bral “visceral larva migrans” syndrome.J Neuroimaging 4:39–40

Cerebral toxocariasis: a possible cause of epileptic seizure in children

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Cerebral toxocariasis: a possible cause of epileptic seizure in children

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