The Current State of Knowledge on Baggio-Yoshinari Syndrome (Brazilian Lyme Disease-like Illness): Chronological Presentation of Historical and Scientific Events Observed over the Last 30 Years.

Baggio-Yoshinari Syndrome (BYS) is an emerging Brazilian tick-borne infectious disease that clinically mimics Lyme Disease (LD) present in the Northern Hemisphere. LD is caused by spirochetes belonging to the Borrelia burgdorferi sensu lato complex and transmitted by Ixodid ticks of complex Ixodes rticinus. On the contrary, BYS is transmitted by hard Ixodid ticks of the genera Amblyomma, Rhipicephalus and Dermacentor. In 1992, the first cases of BYS were described in patients that developed EM rash, flu-like symptoms and arthritis after tick bite episodes. Since these findings, research in BYS has been developing for more than 30 years and shows that its epidemiological, clinical and laboratorial features are different from LD. Borrelia burgdorferi was never isolated in Brazil. In addition, specific serologic tests have shown little positivity. Furthermore, peripheral blood analysis of patients using electron microscopy exhibited structures resembling spirochete-like microorganisms or the latent forms of spirochetes (L form or cell wall deficient bacteria). For these reasons, Brazilian zoonosis was defined as an exotic and emerging Brazilian infectious disease, transmitted by ticks not belonging to the Ixodes ricinus complex, caused by latent spirochetes belonging to the B. burgdorferi sensu lato complex with atypical morphology. The Brazilian ecosystem, combined with its ticks and reservoir biodiversity, possibly contributed to the origin of this new zoonosis, which emerged as a result of the passage of B. burgdorferi through exotic vectors and reservoirs.

Innate and Th1/Th17 adaptive immunity in acute and convalescent Brazilian borreliosis disease.

INTRODUCTION: Brazilian borreliosis (BB) disease is an infectious disease transmitted by ticks that mimics Lyme disease (LD) from the Northern Hemisphere. The BB clinical picture is characterized by a pathognomonic skin lesion (migratory erythema) and joint, neurological, cardiac and psychiatric symptoms. Innate and Th1/Th17 adaptive immunity seem to play an important role in the pathogenesis of Lyme disease. OBJECTIVE: The aim of this study was to characterize the role of innate and Th1/Th17 adaptive immunity in BB patients with acute (<3 months) and convalescent (>3 months) disease. METHODS: Fifty BB patients (28 with acute and 22 with convalescent disease) without treatment and 30 healthy subjects were evaluated. Levels of 20 cytokines or chemokines associated with innate and Th1/Th17 adaptive immunity were analyzed using Luminex (Millipore Corp., Billerica, MA). RESULTS: Overall, BB patients had increased levels of IL-8 (6.29 vs 2.12 p = 0.002) and MIP-1α/CCL3 (5.20 vs 2.06, p = 0.030), associated with innate immunity, and MIP3B/CCL19 (Th1; 297.86 vs 212.41, p = 0.031) and IL-17A (Th17; 3.11 vs 2.20, p = 0.037), associated with adaptive immunity, compared with the levels of healthy controls. When comparing acute BB vs. convalescent BB subjects vs. healthy controls, IL-1ß, IL-8 and MIP-1α/CCL3 (innate mediators) levels were highest in patients in the acute phase of disease (p < 0.05). TNF-α was associated with disseminated symptoms and with humoral reactivity against Borrelia burgdorferi. IL-10 was significantly correlated with IL-6 (r = 0.59, p = 0.003), IL-8 (r = 0.51, p < 0.001), MIP-1α/CCL3 (r = 0.42, p < 0.001) and MIP-3ß/CCL19 (r = 0.40, p = 0.002) in all BB patients. CONCLUSIONS: This is the first study describing that innate and Th1/Th17 adaptive immunity play a crucial role in BB disease. Furthermore, innate mediators are particularly important in acute BB disease, and TNF-α is associated with evolution of BB symptoms.

Innate and Th1/Th17 adaptive immunity in acute and convalescent Brazilian borreliosis disease

Introduction: Brazilian borreliosis (BB) disease is an infectious disease transmitted by ticks that mimics Lyme disease (LD) from the Northern Hemisphere. The BB clinical picture is characterized by a pathognomonic skin lesion (migratory erythema) and joint, neurological, cardiac and psychiatric symptoms. Innate and Th1/Th17 adaptive immunity seem to play an important role in the pathogenesis of Lyme disease. Objective: The aim of this study was to characterize the role of innate and Th1/Th17 adaptive immunity in BB patients with acute (<3 months) and convalescent (>3 months) disease. Methods: Fifty BB patients (28 with acute and 22 with convalescent disease) without treatment and 30 healthy subjects were evaluated. Levels of 20 cytokines or chemokines associated with innate and Th1/Th17 adaptive immunity were analyzed using Luminex (Millipore Corp., Billerica, MA). Results: Overall, BB patients had increased levels of IL-8 (6.29 vs 2.12 p = 0.002) and MIP-1α/CCL3 (5.20 vs 2.06, p = 0.030), associated with innate immunity, and MIP3B/CCL19 (Th1; 297.86 vs 212.41, p = 0.031) and IL-17A (Th17; 3.11 vs 2.20, p = 0.037), associated with adaptive immunity, compared with the levels of healthy controls. When comparing acute BB vs. convalescent BB subjects vs. healthy controls, IL-1β, IL-8 and MIP-1α/CCL3 (innate mediators) levels were highest in patients in the acute phase of disease (p < 0.05). TNF-α was associated with disseminated symptoms and with humoral reactivity against Borrelia burgdorferi. IL-10 was significantly correlated with IL-6 (r = 0.59, p = 0.003), IL-8 (r = 0.51, p < 0.001), MIP-1α/CCL3 (r = 0.42, p < 0.001) and MIP-3β/CCL19 (r = 0.40, p = 0.002) in all BB patients. Conclusions: This is the first study describing that innate and Th1/Th17 adaptive immunity play a crucial role in BB disease. Furthermore, innate mediators are particularly important in acute BB disease, and TNF-α is associated with evolution of BB symptoms.

Innate and Th1/Th17 adaptive immunity in acute and convalescent Brazilian borreliosis disease

ABSTRACT Introduction: Brazilian borreliosis (BB) disease is an infectious disease transmitted by ticks that mimics Lyme disease (LD) from the Northern Hemisphere. The BB clinical picture is characterized by a pathognomonic skin lesion (migratory erythema) and joint, neurological, cardiac and psychiatric symptoms. Innate and Th1/Th17 adaptive immunity seem to play an important role in the pathogenesis of Lyme disease. Objective: The aim of this study was to characterize the role of innate and Th1/Th17 adaptive immunity in BB patients with acute (<3 months) and convalescent (>3 months) disease. Methods: Fifty BB patients (28 with acute and 22 with convalescent disease) without treatment and 30 healthy subjects were evaluated. Levels of 20 cytokines or chemokines associated with innate and Th1/Th17 adaptive immunity were analyzed using Luminex (Millipore Corp., Billerica, MA). Results: Overall, BB patients had increased levels of IL-8 (6.29 vs 2.12 p = 0.002) and MIP-1α/CCL3 (5.20 vs 2.06, p = 0.030), associated with innate immunity, and MIP3B/CCL19 (Th1; 297.86 vs 212.41, p = 0.031) and IL-17A (Th17; 3.11 vs 2.20, p = 0.037), associated with adaptive immunity, compared with the levels of healthy controls. When comparing acute BB vs. convalescent BB subjects vs. healthy controls, IL-1β, IL-8 and MIP-1α/CCL3 (innate mediators) levels were highest in patients in the acute phase of disease (p < 0.05). TNF-α was associated with disseminated symptoms and with humoral reactivity against Borrelia burgdorferi. IL-10 was significantly correlated with IL-6 (r = 0.59, p = 0.003), IL-8 (r = 0.51, p < 0.001), MIP-1α/CCL3 (r = 0.42, p < 0.001) and MIP-3β/CCL19 (r = 0.40, p = 0.002) in all BB patients. Conclusions: This is the first study describing that innate and Th1/Th17 adaptive immunity play a crucial role in BB disease. Furthermore, innate mediators are particularly important in acute BB disease, and TNF-α is associated with evolution of BB symptoms.

Brazilian borreliosis with special emphasis on humans and horses

Abstract Borreliosis caused by Borrelia burgdorferi sensu lato is a cosmopolitan zoonosis studied worldwide; it is called Lyme disease in many countries of the Northern Hemisphere and Lyme-like or Baggio-Yoshinari Syndrome in Brazil. However, despite the increasing number of suspect cases, this disease is still neglected in Brazil by the medical and veterinary communities. Brazilian Lyme-like borreliosis likely involves capybaras as reservoirs and Amblyomma and Rhipicephalus ticks as vectors. Thus, domestic animals can serve as key carriers in pathogen dissemination. This zoonosis has been little studied in horses in Brazil. The first survey was performed in the state of Rio de Janeiro, and this Brazilian Borreliosis exhibits many differences from the disease widely described in the Northern Hemisphere. The etiological agent shows different morphological and genetic characteristics, the disease has a higher recurrence rate after treatment with antibiotics, and the pathogen stimulates intense symptoms such as a broader immune response in humans. Additionally, the Brazilian zoonosis is not transmitted by the Ixodes ricinus complex. With respect to clinical manifestations, Baggio-Yoshinari Syndrome has been reported to cause neurological, cardiac, ophthalmic, muscle, and joint alterations in humans. These symptoms can possibly occur in horses. Here, we present a current panel of studies involving the disease in humans and equines, particularly in Brazil.

Brazilian borreliosis with special emphasis on humans and horses.

Borreliosis caused by Borrelia burgdorferi sensu lato is a cosmopolitan zoonosis studied worldwide; it is called Lyme disease in many countries of the Northern Hemisphere and Lyme-like or Baggio-Yoshinari Syndrome in Brazil. However, despite the increasing number of suspect cases, this disease is still neglected in Brazil by the medical and veterinary communities. Brazilian Lyme-like borreliosis likely involves capybaras as reservoirs and Amblyomma and Rhipicephalus ticks as vectors. Thus, domestic animals can serve as key carriers in pathogen dissemination. This zoonosis has been little studied in horses in Brazil. The first survey was performed in the state of Rio de Janeiro, and this Brazilian Borreliosis exhibits many differences from the disease widely described in the Northern Hemisphere. The etiological agent shows different morphological and genetic characteristics, the disease has a higher recurrence rate after treatment with antibiotics, and the pathogen stimulates intense symptoms such as a broader immune response in humans. Additionally, the Brazilian zoonosis is not transmitted by the Ixodes ricinus complex. With respect to clinical manifestations, Baggio-Yoshinari Syndrome has been reported to cause neurological, cardiac, ophthalmic, muscle, and joint alterations in humans. These symptoms can possibly occur in horses. Here, we present a current panel of studies involving the disease in humans and equines, particularly in Brazil.

Serological evidence of exposure to tick-borne agents in opossums (Didelphis spp. ) in the state of São Paulo, Brazil / Evidência sorológica de exposição a agentes transmitidos por carrapatos em gambás (Didelphis spp. ) no estado de São Paulo, Brasil

Abstract This work involved a serological investigation of tick-borne pathogens in opossums in eight municipalities of the state of São Paulo, Brazil. Serum samples from 109 opossums (91 Didelphis aurita and 18 Didelphis albiventris) were tested to detect antibodies to Rickettsia rickettsii (Taiaçu strain, 1:64 cut-off) and Ehrlichia canis (São Paulo strain, 1:40 cut-off), by indirect immunofluorescence assay (IFA); and against Borrelia burgdorferi (strain G39/40) by enzyme-linked immunosorbent assay (ELISA). The presence of antibodies to anti-R. rickettsii, anti-E. canis and anti-B. burgdorferi was detected in 32 (29.35%), 16 (14.67%) and 30 (27.52%) opossums, respectively. Opossum endpoint titers ranged from 64 to 1,024 for R. rickettsii, from 40 to 160 for E. canis, and from 400 to >51,200 for B. burgdorferi. These serological results suggest that opossums have been exposed to Rickettsia spp., Ehrlichia spp., and B. burgdorferi-related agents in the state of São Paulo. Our study underscores the need for further research about these agents in this study area, in view of the occurrence of Spotted Fever and Baggio-Yoshinari Syndrome disease in humans in the state of São Paulo, Brazil.

Resumo O presente estudo investigou evidência sorológica de agentes transmitidos por carrapatos em gambás em oito municípios do Estado de São Paulo, Brasil. Amostras de soro de 109 gambás (91 Didelphis aurita e 18 Didelphis albiventris) foram testadas para detecção de anticorpos contra Rickettsia rickettsii (cepa Taiaçu, ponto de corte 1:64) Ehrlichia canis (cepa São Paulo, ponto de corte 1:40), pela reação de imunofluorescência indireta (RIFI); e contra Borrelia burgdorferi (cepa G39/40) pelo teste imunoenzimático (ELISA). A presença de anticorpos anti-R. rickettsii, anti-E. canis e anti-B. burgdorferi foi detectada em 32 (29,35%), 16 (14,67%) e 30 (27,52%) gambás, respectivamente. Os títulos finais variaram de 64 a 1.024 para R. rickettsii, de 40 a 160 para E. canis, e de 400 a >51.200 para B. burgdorferi. Esses resultados sugerem que os gambás foram expostos a agentes relacionados à Rickettsia spp., Ehrlichia spp., e B. burgdorferi no Estado de São Paulo. Neste estudo salienta a necessidade de novas pesquisas sobre esses agentes nessas áreas de trabalho, devido à ocorrência da Febre Maculosa e da Síndrome Baggio-Yoshinari em humanos no Estado de São Paulo, Brasil.

Serological evidence of exposure to tick-borne agents in opossums (Didelphis spp.) in the state of São Paulo, Brazil.

This work involved a serological investigation of tick-borne pathogens in opossums in eight municipalities of the state of São Paulo, Brazil. Serum samples from 109 opossums (91 Didelphis aurita and 18 Didelphis albiventris) were tested to detect antibodies to Rickettsia rickettsii (Taiaçu strain, 1:64 cut-off) and Ehrlichia canis (São Paulo strain, 1:40 cut-off), by indirect immunofluorescence assay (IFA); and against Borrelia burgdorferi (strain G39/40) by enzyme-linked immunosorbent assay (ELISA). The presence of antibodies to anti-R. rickettsii, anti-E. canis and anti-B. burgdorferi was detected in 32 (29.35%), 16 (14.67%) and 30 (27.52%) opossums, respectively. Opossum endpoint titers ranged from 64 to 1,024 for R. rickettsii, from 40 to 160 for E. canis, and from 400 to >51,200 for B. burgdorferi. These serological results suggest that opossums have been exposed to Rickettsia spp., Ehrlichia spp., and B. burgdorferi-related agents in the state of São Paulo. Our study underscores the need for further research about these agents in this study area, in view of the occurrence of Spotted Fever and Baggio-Yoshinari Syndrome disease in humans in the state of São Paulo, Brazil.

Anaphylactoid reaction caused by sodium ceftriaxone in two horses experimentally infected by Borrelia burgdorferi.

BACKGROUND: Lyme borreliosis is a disease transmitted by ticks to mammals, especially in horses and humans. Caused by a spirochete Borrelia burgdorferi, it can result in lameness, arthritis, carditis, dermatitis and neurological signs. Anaphylactoid reactions are severe responses caused by direct action of substances (drugs, toxins), which can pose risks to life. Still poorly documented in horses, these reactions are caused by the effects of inflammatory mediators such as histamine, kinins and arachidonic acid metabolites. The last two are the most clinically relevant for the species. CASE PRESENTATION: The simultaneous occurrence of anaphylactoid reaction in two horses experimentally infected by Borrelia burgdorferi undergoing intravenous treatment with ceftriaxone sodium is reported. It was administered 4.7 × 10(8) spirochetes intradermal and subcutaneous applications in both horses to evaluate clinical aspects of the Lyme disease, 95 days before the application of sodium ceftriaxone. During the administration, one horse (a gelding) showed immediate and severe anaphylactoid symptoms such as urticaria, dyspnea, tachycardia, and eyelid edema, which were controlled by injecting dexamethasone. After 1 day, it expressed signs of abdominal discomfort, caused by severe bloat, which was treated surgically via celiotomy. Subsequently, this gelding had piroplasmosis and severe anemia, requiring treatment with an antimicrobial and blood transfusion. Second horse (a mare) showed signs of hypotension during the application of the antibiotic, which disappeared only when the application was interrupted. Days after the event, the mare developed moderate large colon bloat, which was treated with medication only. Subsequently the mare was evolved into the prodromal phase of laminitis in one of the forelimbs, which was treated for 10 days with non-steroidal anti-inflammatory and rheology modifying drugs and cryotherapy. CONCLUSIONS: From the two cases presented here, it does appear that sodium ceftriaxone can induce anaphylactoid reactions in horses infected by Borrelia burgdorferi, which may evolve into colic syndrome, laminitis and the occurrence of opportunistic infections. However, further evidence should be collected in order to draw definite conclusions.

Evidence of Borrelia in wild and domestic mammals from the state of Minas Gerais, Brazil.

The main of the study was to evaluate the presence of Borrelia burgdorferi infection in domestic and wild vertebrates and ectoparasites in endemic areas from the state of Minas Gerais, Brazil. A total of 445 serum samples were examined by ELISA, which used the Borrelia burgdorferi strain G39/40 U.S. source and 3,821 tick samples were tested by polymerase chain reaction (PCR). B. burgdorferi antibodies were found in 30 serum samples (6.74%); three in marsupials (7.69%), three in rodents (2.80%), nine in dogs (6.25%), and 15 in horses (9.68%). Nested-PCR performed in DNA samples obtained from collected ticks demonstrated negative results. Although attempts to amplify B. burgdorferi DNA from ticks had been not successful, the presence of seroreactive vertebrates suggests the possibility the Borrelia species circulating in these regions. Further research is required to provide information on the presence of Borrelia in Brazilian territory and its association with Baggio-Yoshinari syndrome.

First report of mild Brazilian spotted fever associated to arthritis.

We describe the first Brazilian case of mild Rickettsiosis, complicated by knee monoarthritis, in young adult bitten by a tick on his left leg in Camburi zone, located in São Sebastião municipality, southern coastal region of the State of São Paulo, in the Atlantic rainforest region, Brazil. The patient developed inoculation eschar at the tick bite site associated with enlarged lymph nodes in the left groin, fever, polyarthralgia, headache and macular rash. Twenty days after tick bite episode, he displayed monoarthritis in his right knee. The diagnosis of mild Rickettsiosis was established by sequential immunological analysis in serum and synovial fluid, using the indirect immunofluorescence (IF) assay for antibodies reactive with Rickettsia parkeri and Rickettsia rickettsii. The mild Rickettsiosis is an emerging zoonosis, that must be investigated by physicians, including rheumatologists, in patients that present macular rash, fever and eventually arthritis, after visiting the southern coastal Atlantic rainforest region in Brazil.

Chronic lymphomonocytic meningoencephalitis, oligoarthritis and erythema nodosum: report of Baggio-Yoshinari syndrome of long and relapsing evolution.

The Brazilian human borreliosis, also known as Baggio-Yoshinari Syndrome (BYS), is a tickborne disease but whose ticks do not pertain to the Ixodes ricinus complex. It is caused by Borrelia burgdorferi sensu lato microorganisms and resembles clinical and laboratory features of Lyme disease (LD). BYS is also distinguished from LD by its prolonged clinical evolution, with relapsing episodes and autoimmune dysfunction. We describe the case of a young female who, over one year, progressively presented with oligoarthritis, cognitive impairment, menigoencephalitis and erythema nodosum. Diagnosis was established by means of the clinical history and a positive serology to Borrelia burgdorferi sensu strictu. The patient received Ceftriaxone 2 g IV/day during 30 days, followed by 2 months of doxicycline 100 mg bid. Symptoms remitted and the Borrelia serology tests returned to normality. BYS is a new disease described only in Brazil, which has a raising frequency and deserves the attention from the country´s medical board because of clinical, epidemiological and laboratory differences from LD. Despite the fact that it is a hard-to-diagnose zoonosis, it is important to pursuit an early diagnosis because the symptoms respond well to antibiotics or it might be resistant to treatment and may evolve to a chronic phase with both articular and neurological sequelae.

Evidence of Borrelia in wild and domestic mammals from the state of Minas Gerais, Brazil / Evidência de Borrelia em mamíferos silvestres e domésticos no Estado de Minas Gerais, Brasil

The main of the study was to evaluate the presence of Borrelia burgdorferi infection in domestic and wild vertebrates and ectoparasites in endemic areas from the state of Minas Gerais, Brazil. A total of 445 serum samples were examined by ELISA, which used the Borrelia burgdorferi strain G39/40 U.S. source and 3,821 tick samples were tested by polymerase chain reaction (PCR). B. burgdorferi antibodies were found in 30 serum samples (6.74%); three in marsupials (7.69%), three in rodents (2.80%), nine in dogs (6.25%), and 15 in horses (9.68%). Nested-PCR performed in DNA samples obtained from collected ticks demonstrated negative results. Although attempts to amplify B. burgdorferi DNA from ticks had been not successful, the presence of seroreactive vertebrates suggests the possibility the Borrelia species circulating in these regions. Further research is required to provide information on the presence of Borrelia in Brazilian territory and its association with Baggio-Yoshinari syndrome.

O principal objetivo do estudo foi avaliar a presença de infecção por Borrelia burgdorferi em vertebrados domésticos e silvestres e ectoparasitas em áreas endêmicas do estado de Minas Gerais, Brasil. Um total de 445 amostras de soro foram examinadas por ELISA, onde usou-se a cepa americana G39/40 de Borrelia burgdorferi e 3.821 amostras de carrapatos foram testados pela reação em cadeia da polimerase (PCR). Anticorpos anti -B. burgdorferi foram encontrados em 30 amostras de soro (6,74%); três marsupiais (7,69%), três em roedores (2,80%), em nove cães (6,25%) e 15 em cavalos (9,68%). Nested-PCR realizada em amostras de DNA obtidas a partir de carrapatos coletados demonstraram resultados negativos. Apesar das tentativas para amplificar o DNA de B. burgdorferi a partir de carrapatos não tenha sido bem sucedido, a presença de soroatividade em vertebrados sugere a possibilidade de espécies de Borrelia circulando nestas regiões. Mais pesquisas são necessárias para fornecer informações sobre a presença de Borrelia em território brasileiro e sua associação com a Síndrome de Baggio-Yoshinari.

Primeiro caso de febre maculosa brasileira branda associada à artrite / First report of mild Brazilian spotted fever associated to arthritis

Descrevemos o primeiro caso brasileiro de Riquetsiose branda, agravada por monoartrite em joelho, em adulto jovem picado por carrapato na perna esquerda na região de Camburi, localizada no município de São Sebastião, sul da região costeira do estado de São Paulo, Mata Atlântica, Brasil. O paciente apresentou uma escara de inoculação no local da picada do carrapato, associada ao aumento ganglionar em virilha esquerda, febre, poliartralgia, cefaleia e erupção macular. Vinte dias após o episódio da picada de carrapato, o paciente apresentou monoartrite em joelho direito. O diagnóstico de Riquetsiose branda foi estabelecido pela análise imunológica sequencial em amostras de soro e líquido sinovial, tendo sido empregada a técnica de imunofluorescência (IF) indireta para anticorpos reativos contra Rickettsia parkeri e Rickettsia rickettsii. A Riquetsiose branda é uma zoonose emergente, que deve ser investigada pelos médicos, incluindo reumatologistas, em pacientes que apresentem erupção macular, febre e, eventualmente, artrite, após visita ao sul da região costeira da Mata Atlântica no Brasil.

We describe the first Brazilian case of mild Rickettsiosis, complicated by knee monoarthritis, in young adult bitten by a tick on his left leg in Camburi zone, located in São Sebastião municipality, southern coastal region of the State of São Paulo, in the Atlantic rainforest region, Brazil. The patient developed inoculation eschar at the tick bite site associated with enlarged lymph nodes in the left groin, fever, polyarthralgia, headache and macular rash. Twenty days after tick bite episode, he displayed monoarthritis in his right knee. The diagnosis of mild Rickettsiosis was established by sequential immunological analysis in serum and synovial fluid, using the indirect immunofluorescence (IF) assay for antibodies reactive with Rickettsia parkeri and Rickettsia rickettsii. The mild Rickettsiosis is an emerging zoonosis, that must be investigated by physicians, including rheumatologists, in patients that present macular rash, fever and eventually arthritis, after visiting the southern coastal Atlantic rainforest region in Brazil.

Meningoencefalite linfomonocitária crônica, oligoartrite e eritema nodoso: relato de síndrome de Baggio-Yoshinari de longa e recorrente evolução / Chronic lymphomonocytic meningoencephalitis, oligoarthritis and erythema nodosum: report of Baggio-Yoshinari syndrome of long and relapsing evolution

A borreliose humana brasileira, também conhecida como Síndrome de Baggio-Yoshinari (SBY), é uma enfermidade infecciosa própria do território brasileiro, transmitida por carrapatos não pertencentes ao complexo Ixodes ricinus, causada por espiroqueta do gênero Borrelia e que apresenta semelhanças clínicas e laboratoriais com a Doença de Lyme (DL). A SBY distingue-se da DL por apresentar evolução clínica prolongada, com episódios de recorrência e importante disfunção autoimune. Descreveremos o caso de uma paciente jovem, que desenvolveu progressivamente durante cerca de um ano oligoartrite de grandes articulações, seguida de distúrbio do cognitivo, meningoencefalite e eritema nodoso. O diagnóstico foi firmado devido à concomitância de queixas articulares e neurológicas com sorologia positiva para Borrelia burgdorferi sensu stricto. A paciente foi medicada com ceftriaxone 2 g/EV/dia por 30 dias, seguido de dois meses de doxiciclina 100 mg duas vezes ao dia. Houve remissão dos sintomas e normalização dos exames sorológicos para a borreliose. A SBY é uma zoonose emergente descrita apenas no Brasil, cuja frequência tem crescido bastante, e que, em razão das importantes diferenças nos aspectos epidemiológicos, clínicos e laboratoriais em relação à DL, merece especial atenção da classe médica do país. Trata-se de zoonose camuflada e de difícil diagnóstico, mas este deve ser perseguido com tenacidade, pois a enfermidade responde aos antibióticos no estágio inicial, podendo evoluir com sequelas neurológicas e articulares nos casos reconhecidos tardiamente ou recorrentes.

The Brazilian human borreliosis, also known as Baggio-Yoshinari Syndrome (BYS), is a tickborne disease but whose ticks do not pertain to the Ixodes ricinus complex. It is caused by Borrelia burgdorferi sensu lato microorganisms and resembles clinical and laboratory features of Lyme disease (LD). BYS is also distinguished from LD by its prolonged clinical evolution, with relapsing episodes and autoimmune dysfunction. We describe the case of a young female who, over one year, progressively presented with oligoarthritis, cognitive impairment, menigoencephalitis and erythema nodosum. Diagnosis was established by means of the clinical history and a positive serology to Borrelia burgdorferi sensu strictu. The patient received Ceftriaxone 2 g IV/day during 30 days, followed by 2 months of doxicycline 100 mg bid. Symptoms remitted and the Borrelia serology tests returned to normality. BYS is a new disease described only in Brazil, which has a raising frequency and deserves the attention from the country´s medical board because of clinical, epidemiological and laboratory differences from LD. Despite the fact that it is a hard-to-diagnose zoonosis, it is important to pursuit an early diagnosis because the symptoms respond well to antibiotics or it might be resistant to treatment and may evolve to a chronic phase with both articular and neurological sequelae.

Influence of suture on peripheral nerve regeneration and collagen production at the site of neurorrhaphy: an experimental study.

BACKGROUND: Restoration of nerve continuity and effective maintenance of coaptation are considered fundamental principles of end-to-end peripheral nerve repair. OBJECTIVE: To evaluate the influence of the number of stitches on axonal regeneration and collagen production after neurorrhaphy. METHODS: Thirty male Wistar rats were equally divided into 3 groups and were all operated on with the right sciatic nerve exposed. In 2 groups, the nerve was sectioned and repaired by means of 3 (group B) or 6 (group C) epineurium sutures with 10-0 monofilament nylon. One group (group A) was used as a control. Each animal from groups B and C underwent electrophysiological evaluation with motor action potential recordings before nerve section and again at an 8-week interval after neurorrhaphy. Nerve biopsy specimens were used for histomorphometric assessment of axonal regeneration and quantification of collagen at the repair site. RESULTS: Animals from group C had significantly lower motor action potential conduction velocities compared with control animals (P=.02), and no significant difference was seen between groups B and C. Parameters obtained from morphometric evaluation were not significantly different between these 2 groups. Type I collagen and III collagen in the epineurium were significantly higher in group C than in either the control group (P=.001 and P=.003) or group B (P=.01 and P=.02). No differences were identified for collagen I and III in the endoneurium. CONCLUSION: Using 6 sutures for nerve repair is associated with worse electrophysiological outcomes and higher amounts of type I and III collagen in the epineurium compared with control. Neurorraphy with 6 stitches is also related to a significant increase in epineurium collagen I and III compared with 3-stitch neurorraphy.

Profile of patients with Baggio-Yoshinari Syndrome admitted at "Instituto de Infectologia Emilio Ribas" / Perfil dos pacientes com Síndrome Baggio-Yoshinari admitidos no "Instituto de Infectologia Emilio Ribas"

The aim of this study was to evaluate the epidemiological, clinical and laboratorial profile of patients with Baggio-Yoshinari Syndrome (BYS), who underwent internment at the Instituto de Infectologia Emilio Ribas in São Paulo, Brazil, during the period from July 1990 to July 2006. BYS is a new Brazilian tick-borne disease caused by Borrelia burgdorferi sensu lato microorganisms that resembles features of Lyme disease (LD), except for its epidemiological, clinical and laboratorial particularities. From 60 patients' records with positive serology to B. burgdorferi done by ELISA and Western-blotting methods, 19 cases were diagnosed as having BYS, according to criteria adopted at LIM-17 HCFMUSP, the Brazilian Reference Laboratory for the research of BYS. The other 41 remaining patients displayed miscellaneous infections or auto-immune processes. The beginning of symptoms in BYS group varied from one day to six years, from the onset of the disease. Four of 19 patients were included in acute disease stage, and 15 in latent. General unspecific symptoms were identified in almost all cases, with high frequencies of fever (78.9 percent) and lymphadenomegaly (36.8 percent). Six patients had skin lesions (31.5 percent); six arthralgia or arthritis (31.5 percent) and eight neurological symptoms (42 percent). Interestingly, two patients showed antibodies directed to B. burgdorferi exclusively in cerebrospinal fluid. Since BYS is a new emergent Brazilian zoonosis and its diagnosis is sometimes complex, all the new knowledge about BYS must be scattered to Brazilian Medical specialists, aiming to teach them how to diagnose this amazing tick-borne disease and to avoid its progression to chronic irreversible sequels.

O objetivo do estudo foi avaliar o perfil epidemiológico, clínico e laboratorial dos pacientes com Síndrome Baggio-Yoshinari (SBY), internados no Instituto de Infectologia Emilio Ribas, São Paulo, Brasil, no período de julho de 1990 a julho de 2006. SBY é uma nova doença transmitida por carrapatos, causada pela Borrelia burgdorferi sensu lato, que se assemelha a Doença de Lyme (DL), exceto pelas particularidades epidemiológicas, clínicas e laboratoriais. A partir dos registros de 60 pacientes com sorologia positiva para B. burgdorferi pelos métodos de ELISA e Western-blotting, 19 casos foram diagnosticados como SBY, de acordo com critérios adotados pelo LIM-17 HCFMUSP, laboratório de referência para a pesquisa de SBY no Brasil. Os outros 41 pacientes restantes foram classificados como tendo infecções diversas ou processos auto-imunes. O início dos sintomas no grupo SBY variou de um dia a seis anos. Quatro dos 19 pacientes foram incluídos na fase aguda da doença e 15 na fase latente. Os sintomas gerais inespecíficos foram identificados em quase todos os casos, com altas freqüências de febre (78,9 por cento) e linfoadenomegalia (36,8 por cento). Seis pacientes tiveram lesões de pele (31,5 por cento); seis artralgia ou artrite (31,5 por cento) e oito sintomas neurológicos (42 por cento). Curiosamente, dois pacientes apresentaram anticorpos para B. burgdorferi exclusivamente no líquido cefalorraquidiano. Uma vez que a SBY é uma zoonose emergente brasileira e seu diagnóstico é complexo, todo o conhecimento novo sobre SBY deve ser difundido para os médicos brasileiros, com o objetivo de ensiná-los a diagnosticar esta surpreendente doença transmitida por carrapatos, evitando-se a sua progressão para sequelas crônicas irreversíveis.

Growth, cysts and kinetics of Borrelia garinii (Spirochaetales: Spirochaetacea) in different culture media.

The aim of the present paper was to evaluate cyst formation and growth parameters of Borrelia garinii in a range of media differing in formulation and cost. A qualitative assessment of morphology and motility of B. garinii was conducted. All media were prepared aseptically and used in test tubes or Petri dishes. For each medium, the initial spirochete concentration was standardized to 10(3) spirochets/mL. The following culture media were suitable to grow B. garinii: Barbour-Stoenner-Kelly, brain heart infusion and PMR. Growth was minimal at six weeks post-inoculation and maximum spirochete density was observed between 9-12 weeks. Often, the cultures developed cysts of different sizes, isolated or in groups, with a spiraled portion of variable sizes, mainly in unfavorable culture media. Brazilian Lyme disease-like illness, also known as Baggio-Yoshinari syndrome (BYS), is a new and interesting emerging tick-borne disease, caused by Borrelia burgdorferi sensu lato spirochetes, only during its cystic forms. It has been assumed that the peculiar clinical and laboratory features of BYS are consequential to the absence of a human sucker Ixodes ricinus complex tick at risk areas in Brazil, supporting the concept that the borrelia phenotypic expression pattern is modified as it is transmitted through the host.

[Brazilian lyme-like disease or Baggio-Yoshinari syndrome: exotic and emerging Brazilian tick-borne zoonosis].

Lyme disease (LD) is a frequent zoonosis found in the Northern Hemisphere and is considered an infectious disease caused by spirochetes belonging sensu lato to the Borrelia burgdorferi complex transmitted by ticks of the Ixodes ricinus group. In 1992, first cases similar to LD were described in Brazil, when brothers, after a tick bite episode developed symptoms , as erythema migrans, general flu-like symptoms and arthritis. Careful analysis of Brazilian LD-like illness casuistry showed that epidemiological, clinical and laboratorial features in the country were very different from those exhibited by North American and Eurasian LD patients. Human blood-suckers Ixodes ricinus complex ticks were absent at risk areas; the disease is recurrent in the country; Borrelia burgdorferi was never isolated in Brazil and specific serologic tests have shown little positivity with inconsistent results. Furthermore, peripheral blood analysis of patients on electron microscopy exhibited structures resembling Mycoplasma spp, Chlamydia spp and spirochete-like microorganisms. In fact, they were assumed to be latent forms of spirochetes (L form or cell wall deficient bacteria) adapted to survive at inhospitable conditions in vertebrate and invertebrate hosts. For these reasons, the Brazilian zoonosis was named Baggio-Yoshinari Syndrome (BYS) and defined as: "Exotic and emerging Brazilian infectious disease, transmitted by ticks not belonging to the Ixodes ricinus complex, caused by latent spirochetes with atypical morphology, which originates LD-like symptoms, except for occurrence of relapsing episodes and auto-immune disorders".

Growth, cysts and kinetics of Borrelia garinii (Spirochaetales: Spirochaetacea) in different culture media

The aim of the present paper was to evaluate cyst formation and growth parameters of Borrelia garinii in a range of media differing in formulation and cost. A qualitative assessment of morphology and motility of B. garinii was conducted. All media were prepared aseptically and used in test tubes or Petri dishes. For each medium, the initial spirochete concentration was standardized to 10³ spirochets/mL. The following culture media were suitable to grow B. garinii: Barbour-Stoenner-Kelly, brain heart infusion and PMR. Growth was minimal at six weeks post-inoculation and maximum spirochete density was observed between 9-12 weeks. Often, the cultures developed cysts of different sizes, isolated or in groups, with a spiraled portion of variable sizes, mainly in unfavorable culture media. Brazilian Lyme disease-like illness, also known as Baggio-Yoshinari syndrome (BYS), is a new and interesting emerging tick-borne disease, caused by Borrelia burgdorferi sensu lato spirochetes, only during its cystic forms. It has been assumed that the peculiar clinical and laboratory features of BYS are consequential to the absence of a human sucker Ixodes ricinus complex tick at risk areas in Brazil, supporting the concept that the borrelia phenotypic expression pattern is modified as it is transmitted through the host.