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Neurocysticercosis in Infants and Toddlers: Report of Seven Cases and Review of Published Patients
Pediatric Neurology 48 (2013) 432e435
Contents lists available at ScienceDirect
Pediatric Neurology journal homepage: www.elsevier.com/locate/pnu
Original Article
Neurocysticercosis in Infants and Toddlers: Report of Seven Cases and Review of Published Patients Oscar H. Del Brutto MD * School of Medicine, Universidad de Especialidades Espíritu Santo, and Department of Neurological Sciences, Hospital-Clínica Kennedy, Guayaquil, Ecuador
article information
abstract
Article history: Received 19 December 2012 Accepted 5 February 2013
Neurocysticercosis in infants and toddlers has received little attention in the literature, and little is known about the mechanisms of disease acquisition and clinical forms of presentation of the disease in this age group. All patients aged 3 years with neurocysticercosis evaluated at Hospital-Clínica Kennedy, Guayaquil, over a 22-year period were included in this study. Their household contacts were screened to detect Taenia solium carriers, which may represent the source of infection. A literature search on neurocysticercosis in infants and toddlers was also performed to compare personal cases with those described elsewhere. A total of 25 infants and toddlers with neurocysticercosis were included (seven from our institution and 18 from the literature). All patients had seizures as the primary manifestation of the disease, and neuroimaging studies showed one or two parenchymal brain cysticerci in the colloidal stage in 88% of patients. The source of infection was investigated in 11 houses, including the seven households of the present series, and only four of the 18 reported in the literature. A Taenia carrier was found in five (45%) of these households, including three from the present series and two from the literature. A sizable proportion of infants and toddlers with neurocysticercosis have been infected at home. Compulsory search of Taenia carriers among household contacts will allow the detection of the potential source of infection and will reduce further spread of the disease. The search must not be limited to family members, but also extended to domestic employees who are in daily contact with the children. Ó 2013 Elsevier Inc. All rights reserved.
Introduction
Neurocysticercosisddefined as an infection of the central nervous system by the larval stage of the tapeworm Taenia soliumdis the most common helminthic infection of the central nervous system. The disease may occur from infancy to senescence. However, population-based studies performed in disease-endemic communities have shown that children are less often affected than adults [1,2], a percentage that may be even lower among neurocysticercosis patients living in some nonendemic regions such as the United States, Canada, or Western European countries [3-5]. Mechanisms involved in disease acquisition and differences in the reactivity of the immune system * Communications should be addressed to: Dr. Del Brutto; Air Center 3542; PO Box 522970; Miami, FL 33152-2970. E-mail address: [email protected] 0887-8994/$ - see front matter Ó 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.pediatrneurol.2013.02.001
against the parasite may account for the reported lower prevalence of neurocysticercosis in children when compared with that of adults [6]. Taenia solium has a complex lifecycle involving two hosts: humans and pigs. Humans are the only definitive hosts, harboring the adult parasite in the small intestine, whereas both pigs and humans may act as intermediate hosts when they ingest Taenia eggs and develop the larval form called cysticercus. Despite the controversies raised on the form of acquisition of this parasitic disease, recent evidence has made it clear that human cysticercosis is mostly transmitted from person to person, where the source of infection is an asymptomatic Taenia solium carrier [7]. It is also well known that the role of infected swine is to perpetuate the infection lifecycle only. Indeed, due to migration of asymptomatic Taenia carriers, human cysticercosis may occur in regions where swine breeding is nonexistent and in communities where people, for religious reasons, do not eat pork [8,9].
O.H. Del Brutto / Pediatric Neurology 48 (2013) 432e435
Due to limited exposure to external environments, it is likely that younger children have a greater chance of acquiring cysticercosis at home. This could be the case for infants and toddlers; however, that possibility has received little attention in the literature. The primary objective of this study was to better understand the mechanisms involved in disease acquisition. Secondary objectives were to outline the clinical manifestations, imaging findings, and outcomes of neurocysticercosis in patients within this age group (3 years of age). Methods From January 1990 to December 2011, seven patients with neurocysticercosis aged 3 years were evaluated at the Department of Neurological Sciences, Hospital-Clínica Kennedy, Guayaquil (Ecuador). They represented 2% of 346 patients evaluated at this institution during this 22-year period. All patients had definitive diagnoses of neurocysticercosis according to currently accepted diagnostic criteria [10]. All patients and their household contacts were requested to undergo serial coproparasitologic examinations with particular attention to the presence of Taenia eggs, as well as an immunoblot test for the detection of anticysticercal antibodies in serum. Records were reviewed with emphasis on clinical manifestations, neurologic examination, neuroimaging findings, therapy, and outcomes. In addition, a search of the literature on neurocysticercosis in infants and toddlers from 1990 to 2012 was performed using the electronic database of MEDLINE (National Library of Medicine, Bethesda, MD) to compare personal cases with those described elsewhere. For this purpose, the keywords “cysticercosis” and “neurocysticercosis” were combined with “infants,” “toddlers,” “children,” “child,” and “childhood.”
Results Present series (Hospital-Clínica Kennedy)
Five boys and two girls with a mean age of 25.6 9.2 months (age range, 11 to 36 months) were evaluated at the institution during the study years. Five of them belonged to the high-income class of Guayaquil, living in well-urbanized neighborhoods. Seizures were the presenting symptom in all patients, which were simple partial with motor symptomatology in six patients and tonic-clonic generalized in one. Neurologic examination was normal in all but two patients who had transient hemiparesis after a seizure episode. Serum immunoblot for the detection of anticysticercal antibodies was positive in two of six patients in whom the test was performed. All patients were studied with computed tomography (CT), and six of them also underwent magnetic resonance imaging (MRI) of the brain. Neuroimaging studies showed parenchymal brain cysticercosis in all cases, including three patients with a single enhancing lesion (colloidal cysticercus), three with two enhancing lesions, and a single patient with one enhancing lesion plus one cyst (vesicular cysticerci). Sensitivity of CT and MRI was similar, as all lesions found on MRI were also detected by CT. Serial coproparasitologic examinations were negative for Taenia eggs in the seven patients. From a total of 32 household contacts, 26 underwent serial coproparasitologic examinations and 18 had a serum immunoblot. Taenia eggs were found in three patients, including the mother of one patient, and the babysitters of the other two. Serum immunoblot was positive in three additional persons, one of whom was found to have
433
neurocysticercosis on MRI. This was the mother of one of the patients whose babysitter had Taenia eggs. All patients were started on standard regimens of a firstline antiepileptic drug (phenytoin or carbamazepine), and four of them also received a short course of prednisone to reduce brain swelling surrounding the parasites. Albendazole, at daily doses of 15 mg/kg/day for 1 week, was given to three of the four patients aged 24 months (the parents of the other patient refused the use of cysticidal drugs). There were no complications related to therapy in any case. Follow-up for a mean of 87 77 months (range, 6 months to 14 years) was possible in all but one patient who was lost to follow-up. Control neuroimaging studies showed complete resolution of lesions in four cases and residual calcifications in the remaining two. Five of the six patients in whom follow-up was possible remained free of seizures even after the antiepileptic drug was withdrawn, and one patient, who had a residual calcification on control CT, had seizure relapses every time antiepileptic drugs were withdrawn. Literature review
The MEDLINE search revealed 16 articles with reasonably complete information on 20 children aged 3 years with neurocysticercosis [11-26]. Two of these articles (single case reports) were excluded, one because of duplicated publication [11] and the other because it described one of the patients included in the present series [26]. Some other case series of neurocysticercosis in childhood mentioned the occurrence of the disease in infants and toddlers, but specific data were lacking and information could not be abstracted [27-33]. Only two of the 18 infants and toddlers with neurocysticercosis found in the literature were reported from an endemic country (India). The remaining patients were living in nonendemic regions, including the United States (nine cases), countries of the Arabian peninsula (four cases), Western Europe (two cases), and Australia (one case). Two of these 16 patients were born in an endemic country (Ecuador and Congo) and migrated to the nonendemic region before developing the disease, and eight were born of immigrant parents from disease-endemic areas (mainly Latin America). There was no relevant family or travel history in the remaining six autochthonous patients. All patients presented with seizures as the main or sole manifestation of the disease, and the neurologic examination was normal in all but two patients who had focal neurologic signs. Neuroimaging studies revealed one or two parenchymal brain cysts in the colloidal stage (enhancing lesions) in 16 cases and multiple parenchymal brain cysts in the remaining two. None of the patients had hydrocephalus or subarachnoid or ventricular cysts. Serum immunoblot for the detection of anticysticercal antibodies was positive in five of the six patients in whom the test was performed. Serial coproparasitologic examinations of patients and household contacts were performed in only four households, and these were negative for Taenia eggs in four patients but positive in the parents of two of them. All patients received antiepileptic drugs. Specific therapy included the use of albendazole in seven patients and surgical resection of the lesion in a single case. Information
434
O.H. Del Brutto / Pediatric Neurology 48 (2013) 432e435
on outcome was quite partial but, in general terms, the disease followed a benign course in 11 of the 12 patients in whom such information is available. Discussion
This series, together with the literature review, suggests that neurocysticercosis in infants and toddlers most often presents as a relatively benign condition. All patients had seizures as the primary or sole manifestation of the disease, and neuroimaging studies showed one or two parenchymal brain cysticerci in the colloidal stage in 22 (88%) of 25 patients (Fig 1). This presentation of neurocysticercosis is characteristic of patients who do not have contact with infected swine [34]. Indeed, five of seven patients in this study belonged to the high socioeconomic status of Guayaquil, and 16 of the 18 patients reported in the literature came from urban centers of developed countries, where contact with swine is totally absent. Regarding therapy, a few patients received a short course with albendazole, with resolution of lesions on control neuroimaging studies performed a few months after the trial. However, the same results were noticed in patients who received no specific therapy. A total of 18 patients were followed up for a mean of 40 59 months. Of these, only two experienced recurrent seizures when antiepileptic drug therapy was withdrawn. These numbers are too small to conclude on the usefulness or futility of cysticidal drug therapy in infants and toddlers with neurocysticercosis. Moreover, large series of medically treated children (up to 14 years of age) with one or two single enhancing lesions in the brain parenchyma have shown controversial results,
Figure 1. Contrast-enhanced magnetic resonance image showing left and right parietal lobe cysticerci in acute encephalitic phase (on the left).
with some favoring the use of albendazole, while others suggested that the outcome will be the same in patients not receiving specific therapy, but using symptomatic management with corticosteroids alone [35-38]. Due to their limited exposure to external environments, the close surroundings of infants and toddlers with neurocysticercosisdincluding their family members and other household contactsdmust be exhaustively investigated to find the potential source of infection. Although the most reliable examination for these purposes is the immunodiagnosis of Taeniasis by coproantigen detection, this test is not readily available worldwide [39]. So, we must base our search on microscopic stool examinations, which, as known, may face problems related to poor sensitivity and specificity. However, the main problem is not the lack of availability of one test or the lack of reliability of the other, but the lack of knowledge on the basic mechanisms of disease transmission by the attending physicians (particularly those living in nonendemic regions). Moreover, the reviewed literature is plagued with phrases such as “the child did not eat raw pork,” as if that could provide a clue to the diagnosis. Serial coproparasitologic examinations were performed in only four of the 18 households of infants and toddlers with neurocysticercosis reported in the literature. Interestingly, results were positive for Taenia eggs in persons living in two of these houses. In this series, where serial stool examination was requested for all household contacts of infected patients, evidence of persons with Taeniasis was found in three of the seven houses (43%). There are several examples of this form of transmission of cysticercosis (i.e., person to person, from a Taeniasic individual to a healthy child or adult living in the same household). The first of these examples was the occurrence of neurocysticercosis among family members of British soldiers returning home after their duties in India. Those individuals had lived in the United Kingdom and had never traveled abroad. These few cases probably remained unnoticed at a time where the medical attention was focused on the hundreds of British soldiers who developed neurocysticercosis [40]. A more recent and more precisely described example was reported in the Orthodox Jewish community of New York City, where some of their members (child and adults) developed neurocysticercosis as the result of infection from domestic employees who had recently emigrated from Latin America and were found to be Taenia solium carriers, infecting people for whom they worked through nonhygienic handling of food [41]. Indeed the increasing occurrence of neurocysticercosis in traditionally considered nonendemic countries is partly related to the hundreds of Taenia solium carriers entering into these countries every year [42]. Needless to say, the illegal migrational status of many of these persons complicates even more the access to household contacts of neurocysticercosis patients in the search of the Taenia carrier. Physicians evaluating infants and toddlers with neurocysticercosis must be aware of the most common form of disease acquisition in patients from this age group. Compulsory search for Taenia carriers among household contacts of these patients will allow the detection of the potential source of infection and will reduce further spread of the disease to other family members. The search must not
O.H. Del Brutto / Pediatric Neurology 48 (2013) 432e435
be limited to family members, but also include domestic employees who are in daily contact with the children. References [1] Fleury A, Morales J, Bobes RJ, et al. An epidemiological study of familial neurocysticercosis in an endemic Mexican community. Trans R Soc Trop Med Hyg 2006;100(6):551e8. [2] Prasad KN, Verma A, Srivastava S, Gupta RK, Pandey CM, Paliwal VK. An epidemiological study of asymptomatic neurocysticercosis in a pig farming community in northern India. Trans R Soc Trop Med Hyg 2011;105(9):531e6. [3] Serpa JA, White AC Jr. Neurocysticercosis in the United States. Pathog Glob Health 2012;106(5):256e60. [4] Del Brutto OH. A review of cases of human cysticercosis in Canada. Can J Neurol Sci 2012;39(3):319e22. [5] Del Brutto OH. Neurocysticercosis in Western Europe: A reemerging disease? Acta Neurol Belg 2012;112(4):335e43. [6] Sáenz B, Ruíz-Garcia B, Jimenez E, et al. Neurocysticercosis. Clinical, radiologic, and inflammatory differences between children and adults. Pediatr Infect Dis J 2006;25(9):801e3. [7] Lescano AG, Garcia HH, Gilman RH, et al. Taenia solium cysticercosis hotspots surrounding tapeworm carriers: Clustering of human seroprevalence but not on seizures. PLoS Negl Trop Dis 2009;3(1):e371. [8] Leshem E, Kliers I, Bakon M, Gomori M, Karplus R, Schartz E. Neurocysticercosis in travelers: A nation-wide study in Israel. J Travel Med 2011;18(3):191e7. [9] Del Brutto OH. Neurocysticercosis on the Arabian Peninsula, 20032011. Emerg Infect Dis 2013;19(1):172e4. [10] Del Brutto OH, Rajshekhar V, White AC Jr, et al. Proposed diagnostic criteria for neurocysticercosis. Neurology 2001;57(2):177e83. [11] Centers for Disease Control. Locally acquired neurocysticercosisdNorth Carolina, Massachusetts, and South Carolina, 1989-1991. MMWR Morb Mortal Wkly Rep 1992;41(1):1e4. [12] Kruskal BA, Moths L, Teele DW. Neurocysticercosis in a child with no history of travel outside the continental United States. Clin Infect Dis 1993;16(2):290e2. [13] Swischuk LE. Recent onset seizures in infant. Pediatr Emerg Care 1995;11(4):249e51. [14] Stamos JK, Rowley AH, Hahn YS, Chadwick EG, Schantz PM, Wilson M. Neurocysticercosis: report of unusual pediatric cases. Pediatrics 1996;98(5):974e7. [15] Al Shahrani D, Frayha HH, Dabbagh O, Al Shail E. First case of neurocysticercosis in Saudi Arabia. J Trop Pediatr 2003;49(1):58e60. [16] Hussein FMY, Alhajri FA, Buriki KB, El Beltaji AH, Ovais MI, Almuhtaseb S. Neurocysticercosis in Kuwait: Computerized tomography and magnetic resonance imaging findings. Kuwait Med J 2003;35(3):187e91. [17] Wraige E, Graham J, Robb SA, Jan W. Neurocysticercosis masquerading as a cerebral infarct. J Child Neurol 2003;18(4): 298e300. [18] Durá Travé T, Yoldi Petri ME, Bernaloa Iturbe E, Hernández Lagunas T. Neurocisticercosis: Una causa importada de epilepsia sintomática. An Pediatr (Barc) 2003;59(5):500e6. [19] Hira PR, Francis I, Adbella NA, et al. Cysticercosis: Imported and autochthonous infections in Kuwait. Trans R Soc Trop Med Hyg 2004;98(4):233e9. [20] Mody R, Nield LS, Stauffer W, Kamat D. Seizures in a 20-month-old native of Minnesota. Pediatr Emerg Care 2005;21(12):860e2. [21] Scott JX, Devi A, Sathish Kumar T, Moses PD. A rare presentation of neurocysticercosis in a young child. J Trop Pediatr 2005;51(4): 254e5.
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[22] Dhingra B, Mishra D. Disseminated cysticercosis in an infant. Indian Pediatr 2008;45(12):1008e9. [23] Asnis D, Kazakov J, Toronjadze T, et al. Neurocysticercosis in the infant of a pregnant mother with a tapeworm. Am J trop Med Hyg 2009;81(3):449e51. [24] Lucey JM, McCarthy J, Burgner DP. Encysted seizures: status epilepticus in a recently resettled refugee child. Med J Aust 2010; 192(4):237. [25] Khan FY, Imam YZ, Kamel H, Shafaee M. Neurocysticercosis in Qatari patients: Case reports. Travel Med Infect Dis 2011;9(6): 298e302. [26] Del Brutto OH. Neurocysticercosis in a 2-year-old boy infected at home. Pathog Glob Health 2012;106(5):122e3. [27] Rosenfeld EA, Byrd SE, Shulman ST. Neurocysticercosis among children in Chicago. Clin Infect Dis 1996;23(2):262e8. [28] Cuellar R, Molinero M, Ramírez F, Vallejo V. Manifestaciones clínicas de la neurocisticercosis cerebral activa en pediatría. Rev Neurol 1999;29(4):334e7. [29] Morales NMO, Agapejev S, Morales RR, Padula NAMR, Lima MMF. Clinical aspects of neurocysticercosis in children. Pediatr Neurol 2000;22(4):287e91. [30] Pozo-García MP, Campos-Olazábal P, Burneo JG. Neurocisticercosis en una población pediátrica en Lima: Análisis epidemiológico y clínico. Rev Neurol 2003;36(3):205e8. [31] Basu S, Ramchandran U, Thapliyal A. Clinical profile and outcome of pediatric neurocysticercosis. A study from Western Nepal. J Pediatr Neurol 2007;5(1):45e52. [32] Gauchan E, Malla T, Basnet S, Rao KS. Variability of presentations and CT-scan findings in children with neurocysticercosis. Kathmandu Univ Med J 2011;9(34):17e21. [33] Balaji J, Meikandan D. Clinical and radiological profile of neurocysticercosis in South Indian children. Indian J Pediatr 2011;78(8): 1019e20. [34] Del Brutto OH, Nash TE, Garcia HH. Cysticerci-related single parenchymal brain enhancing lesions in non-endemic countries. J Neurol Sci 2012;319(1e2):32e6. [35] Singhi P, Ray M, Singhi S, Khandelwal N. Clinical spectrum of 500 children with neurocysticercosis and response to albendazole therapy. J Child Neurol 2000;15(4):207e13. [36] Gogia S, Talukdar B, Choudhry V, Arora BS. Neurocysticercosis in children: Clinical findings and response to albendazole therapy in a randomized, double-blind, placebo-controlled trial in newly diagnosed cases. Trans R Soc Trop Med Hyg 2003; 97(4):416e21. [37] Kalra V, Dua T, Kumar V. Efficacy of albendazole and short-course dexamethasone treatment in children with 1 or 2 ring-enhancing lesions of neurocysticercosis: A randomized controlled study. J Pediatr 2003;143(1):111e4. [38] Singhi P, Jain V, Khandelwal N. Corticosteroids versus albendazole for treatment of single small enhancing computed tomographic lesions in children with neurocysticercosis. J Child Neurol 2004; 19(5):323e7. [39] Alan JC, Avila G, Garcia Noval J, Flisser A, Craig PS. Immunodiagnosis of taeniasis by coproantigen detection. Parasitology 1990; 101(3):473e7. [40] Dixon HBF, Lipscomb FM. Cysticercosis: An analysis and follow-up of 450 cases. Medical Research Council Special Report Series No 299. London: Her Majesty’s Stationary Office; 1961. [41] Schantz PM, Moore AC, Muñoz JL, et al. Neurocysticercosis in an Orthodox Jewish community in New York City. N Engl J Med 1992; 327(10):692e5. [42] Del Brutto OH, Garcia HH. Neurocysticercosis in nonendemic countries: Time for a reappraisal. Neuroepidemiology 2012;39(2): 145e6.
Contents lists available at ScienceDirect
Pediatric Neurology journal homepage: www.elsevier.com/locate/pnu
Original Article
Neurocysticercosis in Infants and Toddlers: Report of Seven Cases and Review of Published Patients Oscar H. Del Brutto MD * School of Medicine, Universidad de Especialidades Espíritu Santo, and Department of Neurological Sciences, Hospital-Clínica Kennedy, Guayaquil, Ecuador
article information
abstract
Article history: Received 19 December 2012 Accepted 5 February 2013
Neurocysticercosis in infants and toddlers has received little attention in the literature, and little is known about the mechanisms of disease acquisition and clinical forms of presentation of the disease in this age group. All patients aged 3 years with neurocysticercosis evaluated at Hospital-Clínica Kennedy, Guayaquil, over a 22-year period were included in this study. Their household contacts were screened to detect Taenia solium carriers, which may represent the source of infection. A literature search on neurocysticercosis in infants and toddlers was also performed to compare personal cases with those described elsewhere. A total of 25 infants and toddlers with neurocysticercosis were included (seven from our institution and 18 from the literature). All patients had seizures as the primary manifestation of the disease, and neuroimaging studies showed one or two parenchymal brain cysticerci in the colloidal stage in 88% of patients. The source of infection was investigated in 11 houses, including the seven households of the present series, and only four of the 18 reported in the literature. A Taenia carrier was found in five (45%) of these households, including three from the present series and two from the literature. A sizable proportion of infants and toddlers with neurocysticercosis have been infected at home. Compulsory search of Taenia carriers among household contacts will allow the detection of the potential source of infection and will reduce further spread of the disease. The search must not be limited to family members, but also extended to domestic employees who are in daily contact with the children. Ó 2013 Elsevier Inc. All rights reserved.
Introduction
Neurocysticercosisddefined as an infection of the central nervous system by the larval stage of the tapeworm Taenia soliumdis the most common helminthic infection of the central nervous system. The disease may occur from infancy to senescence. However, population-based studies performed in disease-endemic communities have shown that children are less often affected than adults [1,2], a percentage that may be even lower among neurocysticercosis patients living in some nonendemic regions such as the United States, Canada, or Western European countries [3-5]. Mechanisms involved in disease acquisition and differences in the reactivity of the immune system * Communications should be addressed to: Dr. Del Brutto; Air Center 3542; PO Box 522970; Miami, FL 33152-2970. E-mail address: [email protected] 0887-8994/$ - see front matter Ó 2013 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.pediatrneurol.2013.02.001
against the parasite may account for the reported lower prevalence of neurocysticercosis in children when compared with that of adults [6]. Taenia solium has a complex lifecycle involving two hosts: humans and pigs. Humans are the only definitive hosts, harboring the adult parasite in the small intestine, whereas both pigs and humans may act as intermediate hosts when they ingest Taenia eggs and develop the larval form called cysticercus. Despite the controversies raised on the form of acquisition of this parasitic disease, recent evidence has made it clear that human cysticercosis is mostly transmitted from person to person, where the source of infection is an asymptomatic Taenia solium carrier [7]. It is also well known that the role of infected swine is to perpetuate the infection lifecycle only. Indeed, due to migration of asymptomatic Taenia carriers, human cysticercosis may occur in regions where swine breeding is nonexistent and in communities where people, for religious reasons, do not eat pork [8,9].
O.H. Del Brutto / Pediatric Neurology 48 (2013) 432e435
Due to limited exposure to external environments, it is likely that younger children have a greater chance of acquiring cysticercosis at home. This could be the case for infants and toddlers; however, that possibility has received little attention in the literature. The primary objective of this study was to better understand the mechanisms involved in disease acquisition. Secondary objectives were to outline the clinical manifestations, imaging findings, and outcomes of neurocysticercosis in patients within this age group (3 years of age). Methods From January 1990 to December 2011, seven patients with neurocysticercosis aged 3 years were evaluated at the Department of Neurological Sciences, Hospital-Clínica Kennedy, Guayaquil (Ecuador). They represented 2% of 346 patients evaluated at this institution during this 22-year period. All patients had definitive diagnoses of neurocysticercosis according to currently accepted diagnostic criteria [10]. All patients and their household contacts were requested to undergo serial coproparasitologic examinations with particular attention to the presence of Taenia eggs, as well as an immunoblot test for the detection of anticysticercal antibodies in serum. Records were reviewed with emphasis on clinical manifestations, neurologic examination, neuroimaging findings, therapy, and outcomes. In addition, a search of the literature on neurocysticercosis in infants and toddlers from 1990 to 2012 was performed using the electronic database of MEDLINE (National Library of Medicine, Bethesda, MD) to compare personal cases with those described elsewhere. For this purpose, the keywords “cysticercosis” and “neurocysticercosis” were combined with “infants,” “toddlers,” “children,” “child,” and “childhood.”
Results Present series (Hospital-Clínica Kennedy)
Five boys and two girls with a mean age of 25.6 9.2 months (age range, 11 to 36 months) were evaluated at the institution during the study years. Five of them belonged to the high-income class of Guayaquil, living in well-urbanized neighborhoods. Seizures were the presenting symptom in all patients, which were simple partial with motor symptomatology in six patients and tonic-clonic generalized in one. Neurologic examination was normal in all but two patients who had transient hemiparesis after a seizure episode. Serum immunoblot for the detection of anticysticercal antibodies was positive in two of six patients in whom the test was performed. All patients were studied with computed tomography (CT), and six of them also underwent magnetic resonance imaging (MRI) of the brain. Neuroimaging studies showed parenchymal brain cysticercosis in all cases, including three patients with a single enhancing lesion (colloidal cysticercus), three with two enhancing lesions, and a single patient with one enhancing lesion plus one cyst (vesicular cysticerci). Sensitivity of CT and MRI was similar, as all lesions found on MRI were also detected by CT. Serial coproparasitologic examinations were negative for Taenia eggs in the seven patients. From a total of 32 household contacts, 26 underwent serial coproparasitologic examinations and 18 had a serum immunoblot. Taenia eggs were found in three patients, including the mother of one patient, and the babysitters of the other two. Serum immunoblot was positive in three additional persons, one of whom was found to have
433
neurocysticercosis on MRI. This was the mother of one of the patients whose babysitter had Taenia eggs. All patients were started on standard regimens of a firstline antiepileptic drug (phenytoin or carbamazepine), and four of them also received a short course of prednisone to reduce brain swelling surrounding the parasites. Albendazole, at daily doses of 15 mg/kg/day for 1 week, was given to three of the four patients aged 24 months (the parents of the other patient refused the use of cysticidal drugs). There were no complications related to therapy in any case. Follow-up for a mean of 87 77 months (range, 6 months to 14 years) was possible in all but one patient who was lost to follow-up. Control neuroimaging studies showed complete resolution of lesions in four cases and residual calcifications in the remaining two. Five of the six patients in whom follow-up was possible remained free of seizures even after the antiepileptic drug was withdrawn, and one patient, who had a residual calcification on control CT, had seizure relapses every time antiepileptic drugs were withdrawn. Literature review
The MEDLINE search revealed 16 articles with reasonably complete information on 20 children aged 3 years with neurocysticercosis [11-26]. Two of these articles (single case reports) were excluded, one because of duplicated publication [11] and the other because it described one of the patients included in the present series [26]. Some other case series of neurocysticercosis in childhood mentioned the occurrence of the disease in infants and toddlers, but specific data were lacking and information could not be abstracted [27-33]. Only two of the 18 infants and toddlers with neurocysticercosis found in the literature were reported from an endemic country (India). The remaining patients were living in nonendemic regions, including the United States (nine cases), countries of the Arabian peninsula (four cases), Western Europe (two cases), and Australia (one case). Two of these 16 patients were born in an endemic country (Ecuador and Congo) and migrated to the nonendemic region before developing the disease, and eight were born of immigrant parents from disease-endemic areas (mainly Latin America). There was no relevant family or travel history in the remaining six autochthonous patients. All patients presented with seizures as the main or sole manifestation of the disease, and the neurologic examination was normal in all but two patients who had focal neurologic signs. Neuroimaging studies revealed one or two parenchymal brain cysts in the colloidal stage (enhancing lesions) in 16 cases and multiple parenchymal brain cysts in the remaining two. None of the patients had hydrocephalus or subarachnoid or ventricular cysts. Serum immunoblot for the detection of anticysticercal antibodies was positive in five of the six patients in whom the test was performed. Serial coproparasitologic examinations of patients and household contacts were performed in only four households, and these were negative for Taenia eggs in four patients but positive in the parents of two of them. All patients received antiepileptic drugs. Specific therapy included the use of albendazole in seven patients and surgical resection of the lesion in a single case. Information
434
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on outcome was quite partial but, in general terms, the disease followed a benign course in 11 of the 12 patients in whom such information is available. Discussion
This series, together with the literature review, suggests that neurocysticercosis in infants and toddlers most often presents as a relatively benign condition. All patients had seizures as the primary or sole manifestation of the disease, and neuroimaging studies showed one or two parenchymal brain cysticerci in the colloidal stage in 22 (88%) of 25 patients (Fig 1). This presentation of neurocysticercosis is characteristic of patients who do not have contact with infected swine [34]. Indeed, five of seven patients in this study belonged to the high socioeconomic status of Guayaquil, and 16 of the 18 patients reported in the literature came from urban centers of developed countries, where contact with swine is totally absent. Regarding therapy, a few patients received a short course with albendazole, with resolution of lesions on control neuroimaging studies performed a few months after the trial. However, the same results were noticed in patients who received no specific therapy. A total of 18 patients were followed up for a mean of 40 59 months. Of these, only two experienced recurrent seizures when antiepileptic drug therapy was withdrawn. These numbers are too small to conclude on the usefulness or futility of cysticidal drug therapy in infants and toddlers with neurocysticercosis. Moreover, large series of medically treated children (up to 14 years of age) with one or two single enhancing lesions in the brain parenchyma have shown controversial results,
Figure 1. Contrast-enhanced magnetic resonance image showing left and right parietal lobe cysticerci in acute encephalitic phase (on the left).
with some favoring the use of albendazole, while others suggested that the outcome will be the same in patients not receiving specific therapy, but using symptomatic management with corticosteroids alone [35-38]. Due to their limited exposure to external environments, the close surroundings of infants and toddlers with neurocysticercosisdincluding their family members and other household contactsdmust be exhaustively investigated to find the potential source of infection. Although the most reliable examination for these purposes is the immunodiagnosis of Taeniasis by coproantigen detection, this test is not readily available worldwide [39]. So, we must base our search on microscopic stool examinations, which, as known, may face problems related to poor sensitivity and specificity. However, the main problem is not the lack of availability of one test or the lack of reliability of the other, but the lack of knowledge on the basic mechanisms of disease transmission by the attending physicians (particularly those living in nonendemic regions). Moreover, the reviewed literature is plagued with phrases such as “the child did not eat raw pork,” as if that could provide a clue to the diagnosis. Serial coproparasitologic examinations were performed in only four of the 18 households of infants and toddlers with neurocysticercosis reported in the literature. Interestingly, results were positive for Taenia eggs in persons living in two of these houses. In this series, where serial stool examination was requested for all household contacts of infected patients, evidence of persons with Taeniasis was found in three of the seven houses (43%). There are several examples of this form of transmission of cysticercosis (i.e., person to person, from a Taeniasic individual to a healthy child or adult living in the same household). The first of these examples was the occurrence of neurocysticercosis among family members of British soldiers returning home after their duties in India. Those individuals had lived in the United Kingdom and had never traveled abroad. These few cases probably remained unnoticed at a time where the medical attention was focused on the hundreds of British soldiers who developed neurocysticercosis [40]. A more recent and more precisely described example was reported in the Orthodox Jewish community of New York City, where some of their members (child and adults) developed neurocysticercosis as the result of infection from domestic employees who had recently emigrated from Latin America and were found to be Taenia solium carriers, infecting people for whom they worked through nonhygienic handling of food [41]. Indeed the increasing occurrence of neurocysticercosis in traditionally considered nonendemic countries is partly related to the hundreds of Taenia solium carriers entering into these countries every year [42]. Needless to say, the illegal migrational status of many of these persons complicates even more the access to household contacts of neurocysticercosis patients in the search of the Taenia carrier. Physicians evaluating infants and toddlers with neurocysticercosis must be aware of the most common form of disease acquisition in patients from this age group. Compulsory search for Taenia carriers among household contacts of these patients will allow the detection of the potential source of infection and will reduce further spread of the disease to other family members. The search must not
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