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Douglas A. Kerr - One of the best experts on this subject based on the ideXlab platform.
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distinct subtypes of Myelitis in systemic lupus erythematosus
Arthritis & Rheumatism, 2009Co-Authors: Michelle Petri, Julius Birnbaum, Richard E Thompson, Izlem Izbudak, Douglas A. KerrAbstract:Objective Myelitis causes pain, weakness, and sphincteric deficits, and is 1,000-fold more prevalent in patients with systemic lupus erythematosus (SLE) than in the general population. For the last century, descriptions of SLE Myelitis have been primarily limited to case reports. In contrast, larger-scale cohort studies have revealed that Myelitis occurring in the idiopathic demyelinating diseases (i.e., multiple sclerosis versus neuroMyelitis optica) represents distinct syndromes. This study was undertaken to determine whether SLE Myelitis similarly encapsulates distinct syndromes. Methods We analyzed a cohort of 22 patients with SLE and Myelitis. Patients were assessed for neurologic variables related to Myelitis and for clinical and serologic features of SLE. Magnetic resonance images of the spine, cerebrospinal fluid profiles, and autoantibody profiles were obtained. Results Eleven patients presented with signs of gray matter dysfunction (i.e., flaccidity and hyporeflexia), whereas 11 patients presented with signs of white matter dysfunction (i.e., spasticity and hyperreflexia). Patients with gray matter dysfunction were more likely to have irreversible paraplegia (P < 0.01), despite presenting with a monophasic versus polyphasic course (P = 0.01), higher levels of SLE activity (mean SLE Disease Activity Index 9.8 versus 2.0; P = 0.01), and a cerebrospinal fluid profile indistinguishable from bacterial meningitis. Prior to irreversible paraplegia, these patients presented with prodromes of fever and urinary retention, but were misdiagnosed by physicians of different specialties as having urinary tract infections. Patients with white matter dysfunction were more likely to meet criteria for neuroMyelitis optica (P = 0.04) and were also more likely to have antiphospholipid antibodies (lupus anticoagulant) (P = 0.01). Conclusion Our findings indicate that SLE Myelitis encapsulates 2 distinct and previously unrecognized syndromes that can be distinguished clinically by gray matter versus white matter findings. Recognition of fever and urinary retention as prodromes of irreversible paraplegia may allow earlier diagnosis and treatment in SLE patients presenting with gray matter findings.
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Priapism in infantile transverse Myelitis.
Archives of neurology, 2009Co-Authors: Edward R. Hammond, Douglas A. KerrAbstract:Background Transverse Myelitis is an autoimmune neurological disorder of the spinal cord that affects individuals of all age groups, including infants. Objective To report priapism as a unique clinical manifestation of infantile transverse Myelitis. Design Case series. Setting Transverse Myelitis Center at Johns Hopkins, Johns Hopkins Hospital, Baltimore, Maryland. Patients Three infants younger than 12 months. Interventions Intravenous corticosteroids, intravenous immunoglobulin, physical therapy, and rehabilitation. Main Outcome Measure Clinical outcomes in infants with priapism and transverse Myelitis. Results All 3 infants demonstrated rapidly progressive quadriplegia that resulted in substantial disability and proved fatal for 1 infant. Conclusion Priapism in infants with transverse Myelitis may be an indication of severe inflammation and neural injury that necessitates immediate and aggressive treatment.
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Immunopathogenesis of acute transverse Myelitis.
Current opinion in neurology, 2002Co-Authors: Douglas A. Kerr, Harold AyeteyAbstract:Acute transverse Myelitis is a group of disorders characterized by focal inflammation of the spinal cord and resultant neural injury. Acute transverse Myelitis may be an isolated entity or may occur in the context of multifocal or even multisystemic disease. It is clear that the pathological substrate--injury and dysfunction of neural cells within the spinal cord--may be caused by a variety of immunological mechanisms. For example, in acute transverse Myelitis associated with systemic disease (i.e. systemic lupus erythematosus or sarcoidosis), a vasculitic or granulomatous process can often be identified. In idiopathic acute transverse Myelitis, there is an intraparenchymal or perivascular cellular influx into the spinal cord, resulting in the breakdown of the blood-brain barrier and variable demyelination and neuronal injury. There are several critical questions that must be answered before we truly understand acute transverse Myelitis: (1) What are the various triggers for the inflammatory process that induces neural injury in the spinal cord? (2) What are the cellular and humoral factors that induce this neural injury? and (3) Is there a way to modulate the inflammatory response in order to improve patient outcome? Although much remains to be elucidated about the causes of acute transverse Myelitis, tantalizing clues as to the potential immunopathogenic mechanisms in acute transverse Myelitis and related inflammatory disorders of the spinal cord have recently emerged. It is the purpose of this review to illustrate recent discoveries that shed light on this topic, relying when necessary on data from related diseases such as acute disseminated encephaloMyelitis, Guillain-Barre syndrome and neuroMyelitis optica. Developing a further understanding of how the immune system induces neural injury will depend upon confirmation and extension of these findings and will require multicenter collaborative efforts.
Kevin Messacar - One of the best experts on this subject based on the ideXlab platform.
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enterovirus d68 and acute flaccid Myelitis evaluating the evidence for causality
Lancet Infectious Diseases, 2018Co-Authors: Kevin Messacar, Edwin J Asturias, Alison M Hixon, Coretta Van Leerbuter, H G M Niesters, Kenneth L Tyler, Mark J AbzugAbstract:Summary Increased circulation of enterovirus D68 in 2014 and 2016 temporally and geographically coincided with increases in cases of acute flaccid Myelitis, an uncommon condition of paralysis due to lesions in the anterior horn of the spinal cord. The identification of enterovirus D68 in respiratory specimens from cases of acute flaccid Myelitis worldwide further supports an association, yet the absence of direct virus isolation from affected tissues, infrequent detection in cerebrospinal fluid, and the absence, until recently, of an animal model has left the causal nature of the relationship unproven. In this Personal View we evaluate epidemiological and biological evidence linking enterovirus D68 and acute flaccid Myelitis. We applied the Bradford Hill criteria to investigate the evidence for a causal relationship and highlight the importance of comprehensive surveillance and research to further characterise the role of enterovirus D68 in acute flaccid Myelitis and pursue effective therapies and prevention strategies.
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acute flaccid Myelitis a clinical review of us cases 2012 2015
Annals of Neurology, 2016Co-Authors: Kevin Messacar, Kenneth L Tyler, Teri Schreiner, Keith Van Haren, Michele L Yang, Carol A Glaser, Samuel R DominguezAbstract:This review highlights clinical features of the increasing cases of acute flaccid paralysis associated with anterior Myelitis noted in the United States from 2012 to 2015. Acute flaccid Myelitis refers to acute flaccid limb weakness with spinal cord gray matter lesions on imaging or evidence of spinal cord motor neuron injury on electrodiagnostic testing. Although some individuals demonstrated improvement in motor weakness and functional deficits, most have residual weakness a year or more after onset. Epidemiological evidence and biological plausibility support an association between enterovirus D68 and the recent increase in acute flaccid Myelitis cases in the United States. Ann Neurol 2016;80:326-338.
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enterovirus d68 infection in children with acute flaccid Myelitis colorado usa 2014
Emerging Infectious Diseases, 2016Co-Authors: Negar Aliabadi, Kevin Messacar, Daniel M Pastula, Christine C Robinson, Eyal Leshem, James J Sejvar, Steven M Oberste, Daniel R Feikin, Samuel R DominguezAbstract:: During August 8, 2014-October 14, 2014, a total of 11 children with acute flaccid Myelitis and distinctive neuroimaging changes were identified near Denver, Colorado, USA. A respiratory prodrome was experienced by 10, and nasopharyngeal specimens were positive for enterovirus D68 (EV-D68) for 4. To determine whether an association exists between EV-D68 infection and acute flaccid Myelitis, we conducted a retrospective case-control study comparing these patients with 2 groups of outpatient control children (1 group tested for acute respiratory illness and 1 for Bordetella pertussis infection). Adjusted analyses indicated that, for children with acute flaccid Myelitis, the odds of having EV-D68 infection were 10.3 times greater than for those tested for acute respiratory infection and 4.5 times greater than for those tested for B. pertussis infection. No statistical association was seen between acute flaccid Myelitis and non-EV-D68 enterovirus or rhinovirus infection. These findings support an association between EV-D68 infection and acute flaccid Myelitis.
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a novel outbreak enterovirus d68 strain associated with acute flaccid Myelitis cases in the usa 2012 14 a retrospective cohort study
Lancet Infectious Diseases, 2015Co-Authors: Alexander L Greninger, Kevin Messacar, Samia N Naccache, Anna Clayton, Guixia Yu, Sneha Somasekar, Scot Federman, Doug Stryke, Christopher D Anderson, Shigeo YagiAbstract:Summary Background Enterovirus D68 was implicated in a widespread outbreak of severe respiratory illness across the USA in 2014 and has also been reported sporadically in patients with acute flaccid Myelitis. We aimed to investigate the association between enterovirus D68 infection and acute flaccid Myelitis during the 2014 enterovirus D68 respiratory outbreak in the USA. Methods Patients with acute flaccid Myelitis who presented to two hospitals in Colorado and California, USA, between Nov 24, 2013, and Oct 11, 2014, were included in the study. Additional cases identified from Jan 1, 2012, to Oct 4, 2014, via statewide surveillance were provided by the California Department of Public Health. We investigated the cause of these cases by metagenomic next-generation sequencing, viral genome recovery, and enterovirus D68 phylogenetic analysis. We compared patients with acute flaccid Myelitis who were positive for enterovirus D68 with those with acute flaccid Myelitis but negative for enterovirus D68 using the two-tailed Fisher's exact test, two-sample unpaired t test, and Mann-Whitney U test. Findings 48 patients were included: 25 with acute flaccid Myelitis, two with enterovirus-associated encephalitis, five with enterovirus-D68-associated upper respiratory illness, and 16 with aseptic meningitis or encephalitis who tested positive for enterovirus. Enterovirus D68 was detected in respiratory secretions from seven (64%) of 11 patients comprising two temporally and geographically linked acute flaccid Myelitis clusters at the height of the 2014 outbreak, and from 12 (48%) of 25 patients with acute flaccid Myelitis overall. Phylogenetic analysis revealed that all enterovirus D68 sequences associated with acute flaccid Myelitis grouped into a clade B1 strain that emerged in 2010. Of six coding polymorphisms in the clade B1 enterovirus D68 polyprotein, five were present in neuropathogenic poliovirus or enterovirus D70, or both. One child with acute flaccid Myelitis and a sibling with only upper respiratory illness were both infected by identical enterovirus D68 strains. Enterovirus D68 viraemia was identified in a child experiencing acute neurological progression of his paralytic illness. Deep metagenomic sequencing of cerebrospinal fluid from 14 patients with acute flaccid Myelitis did not reveal evidence of an alternative infectious cause to enterovirus D68. Interpretation These findings strengthen the putative association between enterovirus D68 and acute flaccid Myelitis and the contention that acute flaccid Myelitis is a rare yet severe clinical manifestation of enterovirus D68 infection in susceptible hosts. Funding National Institutes of Health, University of California, Abbott Laboratories, and the Centers for Disease Control and Prevention.
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A novel outbreak enterovirus D68 strain associated with acute flaccid Myelitis cases in the USA (2012–14): a retrospective cohort study
The Lancet. Infectious diseases, 2015Co-Authors: Alexander L Greninger, Kevin Messacar, Samia N Naccache, Anna Clayton, Sneha Somasekar, Scot Federman, Doug Stryke, Christopher D Anderson, Shigeo YagiAbstract:Summary Background Enterovirus D68 was implicated in a widespread outbreak of severe respiratory illness across the USA in 2014 and has also been reported sporadically in patients with acute flaccid Myelitis. We aimed to investigate the association between enterovirus D68 infection and acute flaccid Myelitis during the 2014 enterovirus D68 respiratory outbreak in the USA. Methods Patients with acute flaccid Myelitis who presented to two hospitals in Colorado and California, USA, between Nov 24, 2013, and Oct 11, 2014, were included in the study. Additional cases identified from Jan 1, 2012, to Oct 4, 2014, via statewide surveillance were provided by the California Department of Public Health. We investigated the cause of these cases by metagenomic next-generation sequencing, viral genome recovery, and enterovirus D68 phylogenetic analysis. We compared patients with acute flaccid Myelitis who were positive for enterovirus D68 with those with acute flaccid Myelitis but negative for enterovirus D68 using the two-tailed Fisher's exact test, two-sample unpaired t test, and Mann-Whitney U test. Findings 48 patients were included: 25 with acute flaccid Myelitis, two with enterovirus-associated encephalitis, five with enterovirus-D68-associated upper respiratory illness, and 16 with aseptic meningitis or encephalitis who tested positive for enterovirus. Enterovirus D68 was detected in respiratory secretions from seven (64%) of 11 patients comprising two temporally and geographically linked acute flaccid Myelitis clusters at the height of the 2014 outbreak, and from 12 (48%) of 25 patients with acute flaccid Myelitis overall. Phylogenetic analysis revealed that all enterovirus D68 sequences associated with acute flaccid Myelitis grouped into a clade B1 strain that emerged in 2010. Of six coding polymorphisms in the clade B1 enterovirus D68 polyprotein, five were present in neuropathogenic poliovirus or enterovirus D70, or both. One child with acute flaccid Myelitis and a sibling with only upper respiratory illness were both infected by identical enterovirus D68 strains. Enterovirus D68 viraemia was identified in a child experiencing acute neurological progression of his paralytic illness. Deep metagenomic sequencing of cerebrospinal fluid from 14 patients with acute flaccid Myelitis did not reveal evidence of an alternative infectious cause to enterovirus D68. Interpretation These findings strengthen the putative association between enterovirus D68 and acute flaccid Myelitis and the contention that acute flaccid Myelitis is a rare yet severe clinical manifestation of enterovirus D68 infection in susceptible hosts. Funding National Institutes of Health, University of California, Abbott Laboratories, and the Centers for Disease Control and Prevention.
Shigeo Yagi - One of the best experts on this subject based on the ideXlab platform.
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a novel outbreak enterovirus d68 strain associated with acute flaccid Myelitis cases in the usa 2012 14 a retrospective cohort study
Lancet Infectious Diseases, 2015Co-Authors: Alexander L Greninger, Kevin Messacar, Samia N Naccache, Anna Clayton, Guixia Yu, Sneha Somasekar, Scot Federman, Doug Stryke, Christopher D Anderson, Shigeo YagiAbstract:Summary Background Enterovirus D68 was implicated in a widespread outbreak of severe respiratory illness across the USA in 2014 and has also been reported sporadically in patients with acute flaccid Myelitis. We aimed to investigate the association between enterovirus D68 infection and acute flaccid Myelitis during the 2014 enterovirus D68 respiratory outbreak in the USA. Methods Patients with acute flaccid Myelitis who presented to two hospitals in Colorado and California, USA, between Nov 24, 2013, and Oct 11, 2014, were included in the study. Additional cases identified from Jan 1, 2012, to Oct 4, 2014, via statewide surveillance were provided by the California Department of Public Health. We investigated the cause of these cases by metagenomic next-generation sequencing, viral genome recovery, and enterovirus D68 phylogenetic analysis. We compared patients with acute flaccid Myelitis who were positive for enterovirus D68 with those with acute flaccid Myelitis but negative for enterovirus D68 using the two-tailed Fisher's exact test, two-sample unpaired t test, and Mann-Whitney U test. Findings 48 patients were included: 25 with acute flaccid Myelitis, two with enterovirus-associated encephalitis, five with enterovirus-D68-associated upper respiratory illness, and 16 with aseptic meningitis or encephalitis who tested positive for enterovirus. Enterovirus D68 was detected in respiratory secretions from seven (64%) of 11 patients comprising two temporally and geographically linked acute flaccid Myelitis clusters at the height of the 2014 outbreak, and from 12 (48%) of 25 patients with acute flaccid Myelitis overall. Phylogenetic analysis revealed that all enterovirus D68 sequences associated with acute flaccid Myelitis grouped into a clade B1 strain that emerged in 2010. Of six coding polymorphisms in the clade B1 enterovirus D68 polyprotein, five were present in neuropathogenic poliovirus or enterovirus D70, or both. One child with acute flaccid Myelitis and a sibling with only upper respiratory illness were both infected by identical enterovirus D68 strains. Enterovirus D68 viraemia was identified in a child experiencing acute neurological progression of his paralytic illness. Deep metagenomic sequencing of cerebrospinal fluid from 14 patients with acute flaccid Myelitis did not reveal evidence of an alternative infectious cause to enterovirus D68. Interpretation These findings strengthen the putative association between enterovirus D68 and acute flaccid Myelitis and the contention that acute flaccid Myelitis is a rare yet severe clinical manifestation of enterovirus D68 infection in susceptible hosts. Funding National Institutes of Health, University of California, Abbott Laboratories, and the Centers for Disease Control and Prevention.
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A novel outbreak enterovirus D68 strain associated with acute flaccid Myelitis cases in the USA (2012–14): a retrospective cohort study
The Lancet. Infectious diseases, 2015Co-Authors: Alexander L Greninger, Kevin Messacar, Samia N Naccache, Anna Clayton, Sneha Somasekar, Scot Federman, Doug Stryke, Christopher D Anderson, Shigeo YagiAbstract:Summary Background Enterovirus D68 was implicated in a widespread outbreak of severe respiratory illness across the USA in 2014 and has also been reported sporadically in patients with acute flaccid Myelitis. We aimed to investigate the association between enterovirus D68 infection and acute flaccid Myelitis during the 2014 enterovirus D68 respiratory outbreak in the USA. Methods Patients with acute flaccid Myelitis who presented to two hospitals in Colorado and California, USA, between Nov 24, 2013, and Oct 11, 2014, were included in the study. Additional cases identified from Jan 1, 2012, to Oct 4, 2014, via statewide surveillance were provided by the California Department of Public Health. We investigated the cause of these cases by metagenomic next-generation sequencing, viral genome recovery, and enterovirus D68 phylogenetic analysis. We compared patients with acute flaccid Myelitis who were positive for enterovirus D68 with those with acute flaccid Myelitis but negative for enterovirus D68 using the two-tailed Fisher's exact test, two-sample unpaired t test, and Mann-Whitney U test. Findings 48 patients were included: 25 with acute flaccid Myelitis, two with enterovirus-associated encephalitis, five with enterovirus-D68-associated upper respiratory illness, and 16 with aseptic meningitis or encephalitis who tested positive for enterovirus. Enterovirus D68 was detected in respiratory secretions from seven (64%) of 11 patients comprising two temporally and geographically linked acute flaccid Myelitis clusters at the height of the 2014 outbreak, and from 12 (48%) of 25 patients with acute flaccid Myelitis overall. Phylogenetic analysis revealed that all enterovirus D68 sequences associated with acute flaccid Myelitis grouped into a clade B1 strain that emerged in 2010. Of six coding polymorphisms in the clade B1 enterovirus D68 polyprotein, five were present in neuropathogenic poliovirus or enterovirus D70, or both. One child with acute flaccid Myelitis and a sibling with only upper respiratory illness were both infected by identical enterovirus D68 strains. Enterovirus D68 viraemia was identified in a child experiencing acute neurological progression of his paralytic illness. Deep metagenomic sequencing of cerebrospinal fluid from 14 patients with acute flaccid Myelitis did not reveal evidence of an alternative infectious cause to enterovirus D68. Interpretation These findings strengthen the putative association between enterovirus D68 and acute flaccid Myelitis and the contention that acute flaccid Myelitis is a rare yet severe clinical manifestation of enterovirus D68 infection in susceptible hosts. Funding National Institutes of Health, University of California, Abbott Laboratories, and the Centers for Disease Control and Prevention.
Julius Birnbaum - One of the best experts on this subject based on the ideXlab platform.
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distinct subtypes of Myelitis in systemic lupus erythematosus
Arthritis & Rheumatism, 2009Co-Authors: Michelle Petri, Julius Birnbaum, Richard E Thompson, Izlem Izbudak, Douglas A. KerrAbstract:Objective Myelitis causes pain, weakness, and sphincteric deficits, and is 1,000-fold more prevalent in patients with systemic lupus erythematosus (SLE) than in the general population. For the last century, descriptions of SLE Myelitis have been primarily limited to case reports. In contrast, larger-scale cohort studies have revealed that Myelitis occurring in the idiopathic demyelinating diseases (i.e., multiple sclerosis versus neuroMyelitis optica) represents distinct syndromes. This study was undertaken to determine whether SLE Myelitis similarly encapsulates distinct syndromes. Methods We analyzed a cohort of 22 patients with SLE and Myelitis. Patients were assessed for neurologic variables related to Myelitis and for clinical and serologic features of SLE. Magnetic resonance images of the spine, cerebrospinal fluid profiles, and autoantibody profiles were obtained. Results Eleven patients presented with signs of gray matter dysfunction (i.e., flaccidity and hyporeflexia), whereas 11 patients presented with signs of white matter dysfunction (i.e., spasticity and hyperreflexia). Patients with gray matter dysfunction were more likely to have irreversible paraplegia (P < 0.01), despite presenting with a monophasic versus polyphasic course (P = 0.01), higher levels of SLE activity (mean SLE Disease Activity Index 9.8 versus 2.0; P = 0.01), and a cerebrospinal fluid profile indistinguishable from bacterial meningitis. Prior to irreversible paraplegia, these patients presented with prodromes of fever and urinary retention, but were misdiagnosed by physicians of different specialties as having urinary tract infections. Patients with white matter dysfunction were more likely to meet criteria for neuroMyelitis optica (P = 0.04) and were also more likely to have antiphospholipid antibodies (lupus anticoagulant) (P = 0.01). Conclusion Our findings indicate that SLE Myelitis encapsulates 2 distinct and previously unrecognized syndromes that can be distinguished clinically by gray matter versus white matter findings. Recognition of fever and urinary retention as prodromes of irreversible paraplegia may allow earlier diagnosis and treatment in SLE patients presenting with gray matter findings.
Jun-ichi Kira - One of the best experts on this subject based on the ideXlab platform.
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First diagnostic criteria for atopic Myelitis with special reference to discrimination from Myelitis-onset multiple sclerosis.
Journal of the neurological sciences, 2012Co-Authors: Noriko Isobe, Yuji Kanamori, Tomomi Yonekawa, Takuya Matsushita, Hiroshi Shigeto, Nobutoshi Kawamura, Jun-ichi KiraAbstract:To establish the first evidence-based diagnostic criteria for atopic Myelitis (AM) enabling it to be discriminated from Myelitis-onset multiple sclerosis (MS), which is a difficult differential diagnosis. Sixty-nine consecutive AM patients examined from 1996 to 2010 at Kyushu University hospital, who fulfilled the empirical definition of AM (2003), and 51 Myelitis-onset MS patients in whom allergen-specific IgE was measured, were enrolled. The first available brain MRI findings were compared between the two. Then, we compared the clinical and laboratory features between the 16 AM cases who did not meet the Barkhof brain MRI criteria for MS after more than 5 years follow-up and 51 Myelitis-onset MS cases. Based on the discriminative findings, we established diagnostic criteria for AM and calculated the sensitivity and specificity. AM patients had a significantly lower frequency of Barkhof brain lesions on baseline MRI than Myelitis-onset MS patients. AM patients had a significantly higher frequency of present and/or past history of atopic disease and hyperIgEemia, and higher cerebrospinal fluid levels of interleukin 9 and CCL11/eotaxin, but a lower frequency of oligoclonal IgG bands than Myelitis-onset MS patients. Our proposed diagnostic criteria for AM demonstrated 93.3% sensitivity and 93.3% specificity for AM against Myelitis-onset MS, with 82.4% positive predictive value and 97.7% negative predictive value. Our first evidence-based criteria for AM show high sensitivity and specificity, and would be useful clinically. Copyright © 2012 Elsevier B.V. All rights reserved.
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First diagnostic criteria for atopic Myelitis with special reference to discrimination from Myelitis-onset multiple sclerosis
Journal of the Neurological Sciences, 2012Co-Authors: Noriko Isobe, Yuji Kanamori, Tomomi Yonekawa, Takuya Matsushita, Hiroshi Shigeto, Nobutoshi Kawamura, Jun-ichi KiraAbstract:Abstract Objective To establish the first evidence-based diagnostic criteria for atopic Myelitis (AM) enabling it to be discriminated from Myelitis-onset multiple sclerosis (MS), which is a difficult differential diagnosis. Methods Sixty-nine consecutive AM patients examined from 1996 to 2010 at Kyushu University hospital, who fulfilled the empirical definition of AM (2003), and 51 Myelitis-onset MS patients in whom allergen-specific IgE was measured, were enrolled. The first available brain MRI findings were compared between the two. Then, we compared the clinical and laboratory features between the 16 AM cases who did not meet the Barkhof brain MRI criteria for MS after more than 5 years follow-up and 51 Myelitis-onset MS cases. Based on the discriminative findings, we established diagnostic criteria for AM and calculated the sensitivity and specificity. Results AM patients had a significantly lower frequency of Barkhof brain lesions on baseline MRI than Myelitis-onset MS patients. AM patients had a significantly higher frequency of present and/or past history of atopic disease and hyperIgEemia, and higher cerebrospinal fluid levels of interleukin 9 and CCL11/eotaxin, but a lower frequency of oligoclonal IgG bands than Myelitis-onset MS patients. Our proposed diagnostic criteria for AM demonstrated 93.3% sensitivity and 93.3% specificity for AM against Myelitis-onset MS, with 82.4% positive predictive value and 97.7% negative predictive value. Conclusion Our first evidence-based criteria for AM show high sensitivity and specificity, and would be useful clinically.
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distinct csf cytokine chemokine profiles in atopic Myelitis and other causes of Myelitis
Neurology, 2008Co-Authors: M. Tanaka, Yuji Kawano, Hiroyuki Murai, Takuya Matsushita, Takahisa Tateishi, Hirofumi Ochi, Feng-jun Mei, Motozumi Minohara, Jun-ichi KiraAbstract:Background: We reported the emergence of a distinct Myelitis in patients with atopic diathesis (atopic Myelitis [AM]) by a nationwide survey throughout Japan. Similar cases have recently been reported in Caucasians. Pathologic studies of biopsied spinal cord specimens revealed chronic active inflammation with eosinophilic infiltration. Objective: To clarify the cytokine/chemokine alterations in CSF from patients with AM in comparison to other causes of Myelitis. Methods: We measured 27 cytokines, chemokines, and growth factors simultaneously in CSF from 22 patients with AM, 20 with opticospinal multiple sclerosis (OSMS), 11 with HTLV-1–associated myelopathy (HAM), 9 with Sjogren syndrome–related Myelitis (SM), and 20 with other noninflammatory neurologic diseases (OND), using a fluorescent bead-based immunoassay. Results: In patients with AM, CCL11 and interleukin (IL)-9 were significantly increased as compared with patients with OND and other Myelitis while in patients with OSMS interferon-γ and granulocyte-colony stimulating factor levels were significantly higher than in patients with OND and other causes of Myelitis. Significant increase of IL-17 in comparison to patients with OND was found only in patients with OSMS, irrespective of presence or absence of anti-aquaporin-4 (AQP4) antibody. In patients with HAM, CXCL10 and CCL5 were higher than in patients with OND and other Myelitis. In patients with SM, CCL3 and CCL4 were higher than in patients with OND. In patients with AM, CCL11, IL-9, and IL-1 receptor antagonist (IL-1ra) showed positive correlations with the final Kurtzke Expanded Disability Status Scale scores while IL-1ra and IL-12(p70) had positive correlations with disease duration. Conclusion: Intrathecal upregulation of CCL11 and Th2 cytokines is characteristic of atopic Myelitis, which is distinct from interleukin-17/ interferon-γ–related autoimmune condition of opticospinal multiple sclerosis.
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Distinct CSF cytokine/chemokine profiles in atopic Myelitis and other causes of Myelitis.
Neurology, 2008Co-Authors: M. Tanaka, Yuji Kawano, Hiroyuki Murai, Takuya Matsushita, Takahisa Tateishi, Hirofumi Ochi, Feng-jun Mei, Motozumi Minohara, Jun-ichi KiraAbstract:Background: We reported the emergence of a distinct Myelitis in patients with atopic diathesis (atopic Myelitis [AM]) by a nationwide survey throughout Japan. Similar cases have recently been reported in Caucasians. Pathologic studies of biopsied spinal cord specimens revealed chronic active inflammation with eosinophilic infiltration. Objective: To clarify the cytokine/chemokine alterations in CSF from patients with AM in comparison to other causes of Myelitis. Methods: We measured 27 cytokines, chemokines, and growth factors simultaneously in CSF from 22 patients with AM, 20 with opticospinal multiple sclerosis (OSMS), 11 with HTLV-1–associated myelopathy (HAM), 9 with Sjogren syndrome–related Myelitis (SM), and 20 with other noninflammatory neurologic diseases (OND), using a fluorescent bead-based immunoassay. Results: In patients with AM, CCL11 and interleukin (IL)-9 were significantly increased as compared with patients with OND and other Myelitis while in patients with OSMS interferon-γ and granulocyte-colony stimulating factor levels were significantly higher than in patients with OND and other causes of Myelitis. Significant increase of IL-17 in comparison to patients with OND was found only in patients with OSMS, irrespective of presence or absence of anti-aquaporin-4 (AQP4) antibody. In patients with HAM, CXCL10 and CCL5 were higher than in patients with OND and other Myelitis. In patients with SM, CCL3 and CCL4 were higher than in patients with OND. In patients with AM, CCL11, IL-9, and IL-1 receptor antagonist (IL-1ra) showed positive correlations with the final Kurtzke Expanded Disability Status Scale scores while IL-1ra and IL-12(p70) had positive correlations with disease duration. Conclusion: Intrathecal upregulation of CCL11 and Th2 cytokines is characteristic of atopic Myelitis, which is distinct from interleukin-17/ interferon-γ–related autoimmune condition of opticospinal multiple sclerosis.
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atopic Myelitis with focal amyotrophy a possible link to hopkins syndrome
Journal of the Neurological Sciences, 2008Co-Authors: Jun-ichi Kira, Yuji Kawano, Manabu Osoegawa, Noriko Isobe, Futoshi Mihara, Yasumasa Ohyagi, Hiroyuki MuraiAbstract:Abstract Among 22 consecutive patients with Myelitis, of unknown etiology, and atopic diathesis (atopic Myelitis) who from April 2002 to March 2006 had been studied in our clinic, 5 (23%) showed focal amyotrophy in one or two limbs. These 5 patients were subjected to combined clinical, electrophysiological, neuroimaging and immunological studies. Ages were 18 to 58-years-old (average 39). Four showed amyotrophy of unilateral or bilateral upper limbs while one showed amyotrophy in both thighs. All patients showed on-going denervation potentials in the affected muscles, while motor conduction study including F wave was normal except for in one patient who showed prolonged F wave latency in one nerve. Two had localized high signal intensity lesions involving anterior horns on spinal cord MRI and three showed abnormalities suggesting pyramidal tract involvement on motor evoked potentials. All had a present and/or past history of atopic disorders and specific IgE against common environmental allergens, such as mite antigens and cedar pollens, and four showed mild eosinophilia, all of which were compatible with atopic Myelitis. When clinical and laboratory findings were compared between atopic Myelitis with (n = 5) or without focal amyotrophy (n = 17), the former showed a significantly higher frequency of present and past history of asthma (80% vs. 24%, p = 0.0393) and tended to have higher EDSS scores (3.8 ± 1.6 vs. 3.1 ± 1.4). Two patients showed mild to moderate improvements after immunotherapies such as methylprednisolone pulse therapy or plasma exchange, while two recovered with low dose corticosteroids and one without treatment had a gradually progressive course. Although atopic Myelitis preferentially involves the posterior column of the cervical spinal cord, it is possible that anterior horn cells are affected in some cases of atopic Myelitis, especially in patients with asthma. This suggests a possible link between atopic Myelitis and Hopkins syndrome (asthmatic amyotrophy).
