Nested polymerase chain reaction in cerebrospinal fluid for diagnosing spinal cord schistosomiasis: A promising method.

INTRODUCTION: Spinal cord schistosomiasis is a neglected, disabling neurological disease commonly identified in patients from northeast Brazil. The methods currently available for its diagnosis need improvement. PCR in feces and urine is a sensitive diagnostic tool for diagnosis of schistosomiasis, but its value in the cerebrospinal fluid (CSF) is still unknown. OBJECTIVE: The objective of this study was to detect Schistosoma mansoni DNA in CSF from patients with spinal cord schistosomiasis, using the nested PCR (NPCR) assay. METHODS: This was a cross-sectional study carried out from March 2013 to January 2014 at the Aggeu Magalhães Research Center/FIOCRUZ (Pernambuco state, Brazil). NPCR was used to detect Schistosoma mansoni DNA in CSF samples from 20 patients with spinal cord schistosomiasis and 30 controls. RESULTS: NPCR was positive in 16 patients with spinal cord schistosomiasis and none from the control group (sensitivity 80%; specificity 100%, positive predictive value 100%; negative predictive value 88.2%). CONCLUSION: The NPCR technique is highly sensitive and specific for diagnosis of spinal cord schistosomiasis and can be an important diagnostic tool, particularly in cases with negative CSF serology.

Identification and characterization of immune complexes in the cerebrospinal fluid of patients with spinal cord schistosomiasis.

The pathogenesis of neuroschistosomiasis is largely unknown. Available evidence suggests that it depends on the presence of parasite eggs in the nervous tissue and on the host's immune response. We investigated the presence of immune complexes (ICs) in the cerebrospinal fluid (CSF) of four patients with spinal cord schistosomiasis (SCS), and performed their characterization. ICs containing soluble egg antigen of Schistosoma mansoni (SEA) were found in the CSF of all the SCS patients. To our knowledge, this is the first evidence of ICs containing schistosomal antigens in the CSF of patients with SCS. Further studies are necessary to confirm our findings and investigate the possible roles of ICs in the pathogenesis of this disease.

Myeloradiculitis: a rare event in schistosoma infection.

Schistosomiasis a parasitic disease caused by trematodes is widely distributed in (sub-)tropical countries. Depending on the species the infection manifests clinically as gastrointestinal (preferentially Schistosoma mansoni and S. japonicum) or urinary (preferentially S. haematobium) disorders. Here we present an uncommon case of myeloradiculitis leading to bladder palsy and sensory loss at the lower limbs.

Lumbar intramedullary spinal schistosomiasis presenting as progressive paraparesis: case report.

OBJECTIVE AND IMPORTANCE: Blood fluke infection or schistosomiasis affects up to 300 million people worldwide. Although infection of the hepatic and urogenital systems commonly occurs, central nervous system involvement is rare. When presenting in the spinal cord, schistosomiasis can be difficult to diagnose because it can present as mass lesion or transverse myelitis. We describe a patient with lumbar intramedullary spinal schistosomiasis who presented to a tertiary medical center in the United States and discuss the diagnosis and treatment of this rare disease. CLINICAL PRESENTATION: A 25-year-old Brazilian man presented with progressive, subacute, bilateral lower extremity motor and sensory deficits and disturbances in bladder function. Magnetic resonance imaging revealed a diffusely enhancing mass expanding the region of the conus medullaris. INTERVENTION: Laboratory investigation disclosed serum and cerebrospinal fluid eosinophilia. Ultimately, cerebrospinal fluid serology demonstrated the presence of Schistosoma mansoni. The patient was treated with praziquantel and steroids and made a marked recovery. CONCLUSION: The clinical and radiographic manifestations of spinal schistosomiasis can mimic those of intra-axial spinal tumors and transverse myelitis. To avoid unnecessary surgery or delay in treatment, the clinician must have knowledge of this type of presentation. The increasing volume of international travel and high prevalence of the disease worldwide increases the possibility that the practicing neurosurgeon in the United States may encounter this rare but treatable disease.

[Cerebrospinal fluid in the diagnosis of spinal schistosomiasis]. / Líquido cefalorraquidiano no diagnóstico da esquistossomose raquimedular.

Cerebrospinal fluid (CSF) changes in spinal schistosomiasis have been described. Its characteristic features are mild to moderate pleocytosis, presence of eosinophils, slight to moderate protein increase, elevated gamma globulin concentration and a positive immune assay. Nevertheless, these abnormalities are not always present together and therefore difficulties may arise in the assessment of the diagnosis. The purpose of this paper is to evaluate the importance of each CSF alteration concerning the diagnosis in 22 cases of spinal schistosomiasis. According to the results, only 20% of the cases had all the five feature that are considered to be characteristic of spinal schistosomiasis. Abnormal cell count was present in 86%, protein increase in 77.3%, immunoglobulin G increase in 60,8%, eosinophils were present in 36.8% and indirect fluorescent antibody test was positive in 68.2%. In three cases all CSF parameters studied were within the normal limits. As the most specific test among those described was the indirect fluorescent antibody test, it should be regarded for the diagnosis.

Líquido cefalorraquidiano no diagnóstico da esquistossomose raquimedular / Cerebrospinal fluid in the diagnosis of spinal schistosomiasis

As três espécies de esquistossoma podem comprometer o sistema nervoso. O S. mansoni é responsável pela esquistossomose no Brasil, sendo a mielopatia uma forma grave desta helmintose. O propósito deste trabalho é analisar as alterações do líquido cefalorraquidiano (LCR) para dar mais subsídios para o diagnóstico da esquistossomose raquimedular. Fizeram parte deste estudo 22 amostras de LCR de pacientes com esquistossomose espinal. Os resultados das análises destas amostras mostraram que a associação de alterações do LCR com quadro inflamatório e RIFI-IgM positiva ocorreu em 88 por cento dos pacientes, que o eosinófilo esteve presente em apenas 7 amostras (36,8 por cento), e que 3 dos 22 pacientes estudados apresentaram LCR normal. Conclui-se que o exame de LCR é coadjuvante muito útil para o diagnóstico da neuroesquistossomose.

[Myelopathy: clinical considerations and etiological aspects]. / Mielopatias: considerações clínicas e aspectos etiológicos.

We studied 97 inpatients with myelopathy at the University Hospital Lauro Wanderley from 1985 to 2002. Ages ranged from 10 to 78 years and average age was 39.8 years. All patients were examined by neurologist. Cerebrospinal fluid (CSF) was analysed in 44 (45.3%) cases and immunofluorescence reactions and/or ELISA in CSF were made in 32 (32.9%) cases. Radiologic investigation of the vertebral spine and spinal cord of the 32 (32.9%) cases consisted of 8 radiographies, 10 computed tomography and 14 magnetic resonance image. In 41 (42.2%) cases the lesions were situated in the lower dorsal and lumbar level and complete transverse lesion were registred in 61 (62.8%) cases. There was predominance of transverse myelitis (43.2%) and spinal cord compression (15.46%). Spinal cord schistosomiasis was the form more often of the transverse myelitis (13.4%).

Clinical and cerebrospinal fluid (CSF) profile and CSF criteria for the diagnosis of spinal cord schistosomiasis.

OBJECTIVES: To describe the clinical and CSF findings among patients with presumptive neuroschistosomiasis (NS) and to suggest a classification for the CSF diagnosis of presumptive NS. METHOD: The charts of all patients whose CSF exam was performed at the CSF Lab, Jos Silveira Foundation, Salvador, Brazil, from 1988 to 2002 were reviewed. Those with clinically suspected NS whose indirect fluorescent antibody test (IFA) and or hemagglutination-inhibiting antibodies test (HAI) were positive to S. mansoni were identified. RESULTS: Of 377 patients, 67.9% were males; the median age was 36 years (mean 37 + 16 yrs, range 3-82 yrs). The most frequent complaints were paraparesis (59.9%), urinary retention (36.2%), lower limb pain (22.8%). WBC of CSF (count/mm ) was > 4 in 66.0% (mean 83 + 124, median 40, range 4.3-1,100), protein (mg/dl) was > 40 in 84.6% (mean 185 + 519, median 81, range 41-6,800) and eosinophils were present in 46.9%. IFA and HAI were positive in 75.3%. WBC > 4 and presence of eosinophils were associated with IFA and HAI positive (67.3% versus 51.4%, p 0.014; 49.1% versus 23.0%, p 0.0001, respectively) and protein > 40 was not (85.4% versus 77.0%, p 0.09). Presence of WBC > 4, protein > 40 and eosinophils was associated with IFA and HAI positive (71.6% versus 38.2%, p 0.0003) but presence of eosinophils and any other combination of WBC and protein were not. CONCLUSION: NS should be considered as a possible diagnosis in patients who had had contact with schistosome-infected water and present with spinal cord compromising. Presence of IFA and HAI positive to S. mansoni, WBC > 4, protein > 40 and presence of eosinophils in the CSF may be considered as a criterium of highly probable presumptive diagnosis.

[Acute schistosomiasis with brain involvement. Case report]. / Esquistossomose aguda com comprometimento cerebral. Relato de caso.

A case of acute schistosomiasis with magnetic resonance images (MRI) of the brain suggestive of demyelinating lesions, pyramidal disorder in the lower limbs and normal cerebrospinal fluid is presented. Diagnosis could be established by detection of antibodies on blood and cerebrospinal fluid. Schistosoma mansoni involves the spinal cord more often than the brain. Praziquantel associated to prednisolone was effective in this case. There are few reports of brain involvement with S. mansoni, but its prevalence is probably greater. Due to the paucity of studies, its pathophysiology and therapeutics remain to be better clarified. The immune and MRI aspects are emphasized.

Esquistossomose aguda com comprometimento cerebral: relato de caso / Acute schistosomiasis with brain involvement: case report

É relatado um caso de esquistossomose na fase aguda com comprometimento cerebral precoce, síndrome piramidal nos membros inferiores, líquido cefalorraquidiano (LCR) normal e lesoes com padrao desmielinizante na ressonância magnética (RM) encefálica. O diagnóstico foi comprovado através da imunofluorescência indireta para esquistossomose no LCR. O envolvimento encefálico pelo Schistosoma mansoni é menos frequente do que o medular e a resposta ao tratamento com o praziquantel e a prednisolona foi eficaz neste caso. Sao poucos os relatos de neuroesquistossomose encefálica. Devido a esse fato sua fisiopatologia e terapêutica necessitam de melhores estudos. Os aspectos imunológicos e apresentaçao na RM foram enfatizados.

[Mansonic neuroschistosomiasis: clinical, laboratory and therapeutic aspects]. / Neuroesquistossomose mansônica: aspectos clínicos, laboratoriais e terapêuticos.

The authors report 16 patients bearing the meningomyeloradicular form of Mansonic neuroschistosomiasis diagnosed according to clinical, laboratorial and image criteria. Patients have been observed at the Neurology Ambulatory of the Federal University of Bahia, within the period of April/91 to December/93. They have been treated with praziquantel, associated to corticosteroids. The aim has been to evaluate the drug's efficiency and safety in decreasing the neurological signs and symptoms.

Neuroesquistossomose mansônica: aspectos clínicos, laboratoriais e terapêuticos / Mansonic neuroschistosomiasis: clinical, laboratory and therapeutic aspects

Os autores relatam 16 casos de pacientes com forma meningomielorradicular da neuroesquistossomose mansônica, diagnosticados segundo critérios clinicos, laboratoriais e de imagem, acompanhados no Ambulatório de Neurologia-HUPES-UFBA no período de abril/91 a dezembro/93. Eles foram tratados com praziquantel associado a corticoterapia. O objetivo foi avaliar o grau de eficássia e de segurança da droga na regressao dos sinais e sintomas neurológicos.

Neuroesquistossomose / Central nervous system schistosomiasis

O comprometimento neurológico é uma complicação não muito freqüente da esquistossomose. O envolvimento das porções inferiores da medula e da cauda equina é o mais comum. São relatados dois casos em que os pacientes apresentaram instalação subaguda de paraparesia com sorologia positiva para esquistossomose no líquido cefalorraqueano. Tratamento foi efetuado com corticosteróides e oxaminiquine.

The value of an enzyme-linked immunosorbent assay for the diagnosis of schistosomiasis mansoni myeloradiculopathy.

The role of serological tests on cerebrospinal fluid (CSF) in the diagnosis of neuroschistosomiasis has not been fully elucidated; the condition is essentially diagnosed on the basis of circumstantial evidence, which may lead to an erroneous diagnosis, especially in highly endemic areas. We therefore carried out a prospective case-control study in which we compared the concentrations of immunoglobulin G (IgG) specific for schistosome soluble egg antigen (SEA) present in the CSF of 54 patients with schistosomiasis mansoni myeloradiculopathy (SMMR) with those observed in a control group consisting of 41 patients with epidemiological and serological evidence of exposure to schistosomes, and with other neurological disorders that result in mild to moderate impairment of the blood-brain barrier. Anti-SEA IgG was estimated by an enzyme-linked immunosorbent assay. The sensitivity, specificity and positive and negative predictive values were 56%, 95%, 94% and 62% respectively. Likelihood ratios and the corresponding post-test probabilities were determined for 4 levels of anti-SEA IgG in CSF. A value below 0.1 micrograms/mL practically excluded the possibility of SMMR (post-test probability < 5%), a value above 1.4 micrograms/mL practically confirmed the diagnosis of SMMR (post-test probability > 96%), values of 0.1 to 0.5 microgram/mL had no diagnostic value (post-test probability approximately 45%), and values of 0.6 to 1.4 micrograms/mL were useful in some situations (post-test probability approximately 70%). We conclude that the estimation of anti-SEA IgG in the CSF is useful for the diagnosis of SMMR.

[Spinal cord neuroschistosomiasis: clinical and laboratory evaluation of 5 cases]. / Neuroesquistossomose medular. Avaliação clinico-laboratorial de 5 anos.

Schistosomiasis mansoni is an endemic disease in Brazil. Cases of nervous system schistosomiasis have been referred in our medical literature for the past forty years. In this report five cases of schistosomiasis of the spinal cord are studied. A review of the literature has been made and some clinical, laboratory and therapeutical aspects are discussed.

Neuroesquistossomose medular: avaliaçäo clínico-laboratorial de 5 casos / Medular neuroschistosomiosis: clinico-laboratorial evaluation of 5 cases

A incidência da esquistossomose no Brasil continua alta, atingindo cerca de 10% da populaçäo. Desde o primeiro relato da neuroesquistossomose (NE) em nosso país, há mais de 4 décadas, seu comparecimento na literatura nacional tem mostrado acréscimo significativo. O presente trabalho tem como substrato o estudo de 5 casos de NE medular. Estes casos foram avaliados sob o ponto de vista clínico-laboratorial e submetidos a tratamento medicamentoso. Além disso, é nosso propósito incentivar a pesquisa de novos casos, em portadores de quadro sugestivos desta patologia, sobretudo pelos resultados obtidos neste estudo

Neuroesquistossomose em Alagoas / Neuroschistosomiasis in Alagoas

Este trabalho foi realizado no Hospital do Açúcar e Hospital do SESI, em Maceió. Estudamos aproximadamente 100 pacientes que chegaram aos nossos Serviços com manifestaçöes clínicas sugestivas de neuropatias periféricas, discopatias ou comprometimento do sistema nervoso autônomo, com vistas a estabelecer a etiologia das referidas alteraçöes. Identificamos neste grupo 18 pacientes com neuroesquistossomose, confirmados com análise do líquor cefalorraquiano (L.C.R.), exame parasitológico de fezes (E.P.F.) ou biópsia retal (B.R.). Encontramos, além dos quadros já descritos na literatura, casos com diagnóstico prévio de hérnia discal, hiperplasia prostática ou histeria. A evoluçäo clínica e sobretudo liquórica com o tratamento específico (praziquantel ou oxaminiquine) foi mais favorável do que os relatos prévios da literatura. Houve um caso com história pregressa de mielite transversa alguns anos antes, com remissäo total, porém os estudos de potenciais evocados afastaram a possibilidade de esclerose múltipla. Dentre os trabalhos já descritos o nosso se destaca pela alta incidência de esquistossomose como etiologia dos quadros acima descritos

[Schistosomiasis of the central nervous system: report of a case]. / Esquistossomose do sistema nervoso central: relato de um caso.

The neurological involvement in Manson's Schistosomiasis is rarely reported in the literature. The authors describe a case of a 23 years old patient born in São Francisco-MG. She presented with headache, vomiting, fever and stupor. The neurological examination showed stiffness of the neck, paresis with bilateral muscular hypertonia in the upper members, and loss of motricity in inferior members. There was no sign of hepatosplenomegaly. Cerebrospinal fluid examination showed: a decrease in the glucose content (28 mg/dl), increase in the protein content (113 mg/dl) and an increase in cell number (949 cells/mm3) with predominance of neutrophils (70%) and presence of eosinophils (9%); the indirect immunofluorescence test using samples of mouse's liver with periovular granuloma was reactive (IgG 1/16; IgM 1/4); the search for acid-fast bacilli and fungi, as well as the culture for acid fast bacilli (after 60 days) and the cysticercosis immunological tests were negative. The computerized tomography of the brain and spinal cord did not show abnormalities. Fecal smear examination showed mature eggs of S. mansoni. Treatment was made with corticosteroids and praziquantel (60 mg/kg/day; 7 days), and the cerebrospinal fluid alterations remitted. However, severe neurological sequels (paraplegia and neurogenic bladder) remained.

Esquistossomose do sistema nervoso central: relato de um caso / Schistosomiasis of the central nervous system: report of a case

O comprometimento do sistema nervoso na esquistossomose mansônica é evento raramente diagnosticado na evoluçäo desta parasitose. Os autores descrevem o caso de uma paciente de 23 anos, natural de Säo Francisco-MG, que apresentava há duas semanas quadro de cefaléia, vômitos em jato, febre e sonolência: o exame neurológico mostrava rigidez de nuca, paresta com hipertonia muscular bilateral em membros superiores, perda de motricidade e sensibilidade em membros inferiores. Näo se palpava hepato-esplenomegalia. O exame do líquido cefalorraquidiano (LCR) mostrou hipoglicorraqui (28 mg%), hiperproteinorraquia (113 mg%) e pleocitose acentuada (949 células/mm**3), com predomínio de neutrófilos (70%), além de eosinofilorraquia (9%). A reaçäo de imunofluorescência indireta para esquistossomose, no LCR, realizada em cortes de fígado com granuloma peri-ovular foi reagente (IgG: 1/16; IgM: 1/4). As pesquisas de fungos e b.a.a.r. no LCR, assim como a cultura para b.a.a.r. (apóos 60 dias) e as reaçöes imunológicas para neurocisticercose foram negativas. Uma tomografia computadorizada do cérebro e medula espinhal näo evidenciou anormalidades. No exame parasitológico das fezes foi identificada a presença de ovos viáveis de S. mansoni. Instituiu-se tratamento com corticóides e praziquantel (60 mg/kg/dia; 7 dias) tendo havido regressäo das alteraçöes do LCR, permanecendo entretanto seqüelas neurológicas graves (paraplegia e bexiga neurogênica)