Retrospective analysis of long-term outcome of chronic progressive neurological manifestations in Behcet's disease

Hirohata, Shunsei; Kikuchi, Hirotoshi; Sawada, Tetsuji; Nagafuchi, Hiroko; Kuwana, Masataka; Takeno, Mitsuhiro; Ishigatsubo, Yoshiaki

doi:10.1016/j.jns.2015.01.005

Cited by 36 publications

(21 citation statements)

References 20 publications

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“…The use of corticosteroids to treat neurologic diseases is controversial. In human medicine, several studies have failed to show a beneficial or a deleterious effect of corticosteroids in patients with infectious or traumatic meningitis, or both, suggesting the presence of confounding factors. For example, the use of corticosteroids is recommended for treatment of Haemophilus influenza meningitis in children, but evidence for beneficial or deleterious effects in adults for tuberculous meningitis is lacking .…”

Section: Discussionmentioning

confidence: 99%

Cerebrospinal Nematodiasis in 20 Camelids

Bertin

Taylor

2016

Veterinary Internal Medicne

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BackgroundInformation about the clinical and clinicopathologic aspects of cerebrospinal nematodiasis (CN) in camelids is limited.HypothesisClinical and therapeutic variables will be identified as factors predictive of survival.AnimalsClient‐owned camelids suspected of having CN admitted to Purdue University between 1995 and 2015.MethodsA retrospective study was performed. A diagnosis of CN was based on cerebrospinal fluid (CSF) eosinophilic pleocytosis or postmortem findings.ResultsEleven alpacas and 9 llamas met the inclusion criteria. Seventy‐five percent of the camelids were male (27% castrated and 73% intact). Common clinical abnormalities included proprioceptive deficits (100% of animals), recumbency (55%), tachypnea (55%), and ataxia (40%). Among the 85% of treated animals, 100% received PO fenbendazole, and 88% received a nonsteroidal anti‐inflammatory drug. The survival rate to discharge was 45%. Plasma fibrinogen concentration, creatine kinase activity, and serum creatinine concentration were significantly higher in nonsurvivors. Blood eosinophil count, platelet count, and total CO 2 were significantly lower in nonsurvivors. Factors associated with survival were species, sex, absence of treatment with corticosteroids, and clinical improvement. There was no association between recumbency at admission and survival. A plasma fibrinogen concentration above >266 mg/dL was an excellent diagnostic test to predict survival in the presence of neurological signs or CSF eosinophilia.ConclusionsAlthough prognosis for CN in camelids is guarded, presence of recumbency at admission is not predictive of nonsurvival. Male camelids and llamas appear more likely to die from CN. Corticosteroid treatment is contraindicated in animals diagnosed with CN.

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Section: Discussionmentioning

confidence: 99%

Cerebrospinal Nematodiasis in 20 Camelids

Bertin

Taylor

2016

Veterinary Internal Medicne

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“…In contrast, chronic progressive NBD is characterized by intractable slowly progressive dementia, ataxia and dysarthria with persistent elevation of the CSF IL-6 and progressive brain stem atrophy on MRI [85]. Acute NBD responds well to high doses of corticosteroid, while chronic progressive NBD does not respond to steroid, but dose respond to low doses of methotrexate [86]. The histopathological findings of NBD from biopsied and autopsied brain sections have shown the infiltration of inflammatory cells around small vessels with neuron loss, particular in the brainstem [82].…”

Section: Neuro-behçet Diseasementioning

confidence: 99%

Blood–brain barrier dysfunction in immuno-mediated neurological diseases

Shimizu

Nishihara

Kanda

2018

Immunological Medicine

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The blood-brain barrier (BBB) is the brain-specific endothelial cell barrier that is important for maintaining brain homeostasis and preventing the entry of toxic substances. Pathological BBB dysfunction is a critical step of the disease process in several immuno-mediated neurological diseases, including multiple sclerosis (MS), neuromyelitis optica (NMO), neuropsychiatric systemic lupus erythematosus (NPSLE) and neuro-Behçet diseases. The pathological findings from patients with secondary progressive (SP) MS, NMO and NPSLE showed leaky BBB in the active lesions. NMO is a disease with strong evidence of disease-specific and pathogenic autoantibodies (aquaporin 4 [AQP4] autoantibodies). In the development of NMO, circulating AQP4 autoantibodies need to pass through the BBB in order to reach AQP4 on the astrocyte endfeet. Strong evidence suggests that NPSLE is associated with the disruption of the BBB and NPSLE patients frequently have antibodies bound to endothelial cells in their sera. We recently identified two BBB-reactive autoantibodies in immuno-mediated neurological diseases: galectin-3 autoantibodies in SPMS and GRP78 autoantibodies in NMO. In the present review article, we describe the basic structure and cellular biology of the BBB, discuss recent insights regarding the pathophysiology of the BBB breakdown in the setting of immuno-mediated neurological diseases, and describe our recent findings of autoantibody-mediated BBB breakdown.

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“…Indeed, clinical manifestations of A-P-NBD subside in response to moderate to high dose of corticosteroid and the relapse is significantly suppressed by colchicine 34 . On the other hand, few studies have shown therapeutic effects of corticosteroid and conventional immunosuppressants on CP-P-NBD except methotrexate 35 . Methotrexate is recommended as the first line therapy for CP-P-NBD in the guidelines for management of NBD from Behcet’s Disease Research Committee, Ministry of Health, Welfare, and Labour, Japan.…”

Section: Discussionmentioning

confidence: 98%

Distinct clinical features between acute and chronic progressive parenchymal neuro-Behçet disease: meta-analysis

Ishido

Horita

Takeuchi

et al. 2017

Sci Rep

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Neuro-Behçet’s disease (NBD) is subcategorized into parenchymal-NBD (P-NBD) and non-parenchymal-NBD types. Recently, P-NBD has been further subdivided into acute P-NBD (A-P-NBD) and chronic progressive P-NBD (CP-P-NBD). Although an increasing number of studies have reported the various clinical features of A-P-NBD and CP-P-NBD over the last two decades, there was a considerable inconsistency. Two investigators systematically searched four electrical databases to detect studies that provided sufficient data to assess the specific characteristics of A-P-NBD and CP-P-NBD. All meta-analysis was carried out by employing the random-model generic inverse variance method. We included 11 reports consisted of 184 A-P-NBD patients and 114 CP-P-NBD patients. While fever (42% for A-P-NBD, 5% for CP-P-NBD, p < 0.001, I2 = 93%) was more frequently observed in A-P-NBD cases; sphincter disturbances (9%, 34%, P = 0.005, I2 = 87%), ataxia (16%, 57%, P < 0.001, I2 = 92%), dementia (7%, 61%, P < 0.001, I2 = 97%), confusion (5%, 18%, P = 0.04, I2 = 76%), brain stem atrophy on MRI (4%, 75%, P < 0.001, I2 = 98%), and abnormal MRI findings in cerebellum (7%, 54%, P = 0.02, I2 = 81%) were more common in CP-P-NBD. Cerebrospinal fluid cell count (94/mm3, 11/mm3, P = 0.009, I2 = 85%) was higher in A-P-NBD cases. We demonstrated that A-P-NBD and CP-P-NBD had clearly different clinical features and believe that these data will help future studies investigating P-NBD.

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Retrospective analysis of long-term outcome of chronic progressive neurological manifestations in Behcet's disease

Cited by 36 publications

References 20 publications

Cerebrospinal Nematodiasis in 20 Camelids

Cerebrospinal Nematodiasis in 20 Camelids

Blood–brain barrier dysfunction in immuno-mediated neurological diseases

Distinct clinical features between acute and chronic progressive parenchymal neuro-Behçet disease: meta-analysis

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