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opsoclonus myoclonus syndrome

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https://www.readbyqxmd.com/read/29135842/genomic-profiles-of-neuroblastoma-associated-with-opsoclonus-myoclonus-syndrome
#1
Barbara Hero, Nathalie Clement, Ingrid Øra, Gaelle Pierron, Eve Lapouble, Jessica Theissen, Claudia Pasqualini, Dominique Valteau-Couanet, Dominique Plantaz, Jean Michon, Olivier Delattre, Marc Tardieu, Gudrun Schleiermacher
Opsoclonus myoclonus syndrome (OMS), often called "dancing eyed syndrome," is a rare neurological condition associated with neuroblastoma in the majority of all childhood cases. Genomic copy number profiles have shown to be of prognostic significance for neuroblastoma patients. The aim of this retrospective multicenter study was to analyze the genomic copy number profiles of tumors from children with neuroblastoma presenting with OMS at diagnosis. In 44 cases of neuroblastoma associated with OMS, overall genomic profiling by either array-comparative genomic hybridization or single nucleotide polymorphism array proved successful in 91% of the cases, distinguishing tumors harboring segmental chromosome alterations from those with numerical chromosome alterations only...
November 13, 2017: Journal of Pediatric Hematology/oncology
https://www.readbyqxmd.com/read/29121544/brain-volumetric-analysis-and-cortical-thickness-in-adults-with-saccadic-intrusions-ocular-flutter-or-opsoclonus-myoclonus-syndrome
#2
María-José Ibáñez-Juliá, Evangelia Pappa, Bertrand Gaymard, Delphine Leclercq, Charlotte Hautefort, Caroline Tilikete, Jean-Yves Delattre, Khê Hoang-Xuan, Dimitri Psimaras, Agusti Alentorn
OBJECTIVES: Ocular flutter (OF) and opsoclonus are considered a continuum with a similar pathogenesis. Due to the rarity of this disease in the adult population, little is known about the brain morphological changes in the chronic phase of the disease. PATIENTS AND METHODS: Six magnetic resonance imaging from adults with previous history of OF/Opsoclonus and 12 healthy patients (paired by age and sex) were analyzed in order to identify the long term cortical thickness pattern in this rare disease by using Freesurfer...
October 31, 2017: Clinical Neurology and Neurosurgery
https://www.readbyqxmd.com/read/29103425/cancer-and-autoimmunity-paraneoplastic-neurological-disorders-associated-with-neuroblastic-tumors
#3
Wendy G Mitchell, Franz Blaes
Cancer and autoimmunity come together in paraneoplastic syndromes (PNS), which reflect the remote, not direct, effects of cancer. In the pediatric population, a variety of PNS have been described, but the most common of these rare disorders are instigated by neuroblastic tumors, such as neuroblastoma, ganglioneuroblastoma, and ganglioneuroma. The main pediatric-onset neurological PNS are ROHHAD syndrome, anti-ANNA1 (anti-Hu), and opsoclonus-myoclonus syndrome. They manifest distinctive neurological features, which aid the diagnosis, though under-recognition still poses serious challenges and risks...
August 2017: Seminars in Pediatric Neurology
https://www.readbyqxmd.com/read/29097081/paraneoplastic-autoimmune-movement-disorders
#4
Thien Thien Lim
PURPOSE OF REVIEW: To provide an overview of paraneoplastic autoimmune disorders presenting with various movement disorders. RECENT FINDINGS: The spectrum of paraneoplastic autoimmune disorders has been expanding with the discovery of new antibodies against cell surface and intracellular antigens. Many of these paraneoplastic autoimmune disorders manifest as a form of movement disorder. With the discovery of new neuronal antibodies, an increasing number of idiopathic or neurodegenerative movement disorders are now being reclassified as immune-mediated movement disorders...
October 13, 2017: Parkinsonism & related Disorders
https://www.readbyqxmd.com/read/29058581/opsoclonus-myoclonus-syndrome-in-a-patient-with-japanese-encephalitis-a-case-report
#5
Subatharshini Sountharalingam, H M M T B Herath, Dharshana Wijegunasinghe, Sunethra Senanayke
BACKGROUND: Opsoclonus myoclonus syndrome is a rare neurological disorder that usually manifests as a paraneoplastic phenomenon. Although some viruses are reported to cause this condition, opsoclonus myoclonus syndrome by Japanese encephalitis has not been reported previously. CASE PRESENTATION: Here we present the case of a 31-year-old Sri Lankan woman who presented with fever, altered level of consciousness, opsoclonus, and facial myoclonus. She was diagnosed as having Japanese encephalitis based on cerebrospinal fluid and serum Japanese encephalitis-specific immunoglobulin M antibody and characteristic magnetic resonance imaging abnormalities...
October 23, 2017: Journal of Medical Case Reports
https://www.readbyqxmd.com/read/29017268/the-spectrum-and-burden-of-influenza-associated-neurological-disease-in-children-combined-encephalitis-and-influenza-sentinel-site-surveillance-from-australia-2013-2015
#6
Philip N Britton, Christopher C Blyth, Kristine Macartney, Russell C Dale, Jean Li-Kim-Moy, Gulam Khandaker, Nigel W Crawford, Helen Marshall, Julia E Clark, Elizabeth J Elliott, Robert Booy, Allen C Cheng, Cheryl A Jones
Background: There are few longitudinal studies of seasonal influenza-associated neurological disease (IAND) and none from the Southern Hemisphere. Methods: We extracted prospectively acquired Australian surveillance data from 2 studies nested within the Paediatric Active Enhanced Disease Surveillance (PAEDS) network: the Influenza Complications Alert Network (FluCAN) study and the Australian Childhood Encephalitis (ACE) study between 2013 and 2015. We described the clinical features and severity of IAND in children, including influenza-associated encephalitis/encephalopathy (IAE)...
August 15, 2017: Clinical Infectious Diseases: An Official Publication of the Infectious Diseases Society of America
https://www.readbyqxmd.com/read/28991699/microglial-macrophage-markers-chi3l1-scd14-and-scd163-in-csf-and-serum-of-pediatric-inflammatory-and-non-inflammatory-neurological-disorders-a-case-control-study-and-reference-ranges
#7
Michael R Pranzatelli, Elizabeth D Tate, Nathan R McGee
OBJECTIVE: To assess the role of microglia and macrophages in neuroinflammatory disorders in children via biomarkers, and establish control reference ranges. METHODS: In an IRB-approved case-control study of 98 children, the concentrations of CSF/serum CHI3L1, sCD14, and sCD163 were measured by ELISA. Groups were controls (non-inflammatory neurological disorders, NIND, n=37), opsoclonus-myoclonus syndrome (OMS, n=37), and other inflammatory neurological disorders (OIND, n=24)...
October 15, 2017: Journal of the Neurological Sciences
https://www.readbyqxmd.com/read/28975137/rapid-resolution-of-enterovirus-71-associated-opsoclonus-myoclonus-syndrome-on-intravenous-immunoglobulin
#8
Ahmed Sahly, Laurence Gauquelin, Guillaume Sébire
Nonparaneoplastic opsoclonus-myoclonus ataxia syndrome is a rare neuroinflammatory condition featured by opsoclonus, myoclonus, ataxia, and cognitive behavioral disturbance. The authors report an observation of enterovirus 71-associated opsoclonus-myoclonus ataxia syndrome evolving toward full recovery on intravenous intravenous immunoglobulin (IG) treatment. Based on this case report, enterovirus 71 should be added to the list of infectious agents likely involved in opsoclonus-myoclonus ataxia syndrome, including the emerging subgroup of opsoclonus-myoclonus ataxia syndrome recovering without aggressive or prolonged immunosuppressive intervention...
January 2017: Child neurology open
https://www.readbyqxmd.com/read/28971571/clinicopathological-features-of-neuroblastic-tumors-with-opsoclonus-myoclonus-ataxia-syndrome-follicular-structure-predicts-a-better-neurological-outcome
#9
Hiroko Fukushima, Takeshi Inoue, Yuichi Takama, Naomi Ishii, Takahiro Okuno, Yasutsugu Kobayashi, Akihiro Yoneda, Tetsuro Nakamura, Ichiro Kuki, Junichi Hara
Neuroblastic tumors (NT) with opsoclonus-myoclonus syndrome (OMS) display characteristic histological features, such as lymphocytic infiltration with lymphoid follicles, indicating an underlying immune response. We retrospectively assessed NT patients from 2001 to 2016. Five cases of NT with OMS and 76 cases of NT without OMS were histopathologically reviewed in this study. The grade of lymphocytic infiltration was evaluated. The number of follicles was counted and the presence or absence of lymphoid follicles was recorded for each case...
October 2017: Pathology International
https://www.readbyqxmd.com/read/28959231/demographic-clinical-and-immunologic-features-of-389-children-with-opsoclonus-myoclonus-syndrome-a-cross-sectional-study
#10
Michael R Pranzatelli, Elizabeth D Tate, Nathan R McGee
Pediatric-onset opsoclonus-myoclonus syndrome (OMS) is a devastating neuroinflammatory, often paraneoplastic, disorder. The objective was to characterize demographic, clinical, and immunologic aspects in the largest cohort reported to date. Cross-sectional data were collected on 389 children in an IRB-approved, observational study at the National Pediatric Myoclonus Center. Non-parametric statistical analysis was used. OMS manifested in major racial/ethnic groups, paralleling US population densities. Median onset age was 1...
2017: Frontiers in Neurology
https://www.readbyqxmd.com/read/28926689/therapeutic-plasma-exchange-for-a-case-of-refractory-opsoclonus-myoclonus-ataxia-syndrome
#11
Jocelyn E Greensher, James Louie, Jonathan D Fish
Opsoclonus myoclonus ataxia syndrome (OMAS) can be refractory to standard therapies and devastating. Alternative treatments are imperative. A 14-month-old male diagnosed with neuroblastoma and paraneoplastic OMAS achieved complete cancer remission with chemotherapy. The OMAS, however, persisted over the subsequent 4 years despite numerous immune-modulatory and immunosuppressive therapies. The patient ultimately achieved complete remission following therapeutic plasma exchange (TPE) combined with rituximab and intravenous immunoglobulin...
September 19, 2017: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/28905654/opsoclonus-myoclonus-syndrome-following-long-term-use-of-cyclosporine
#12
Hyun Goo Kang, Sung Kwang Park, Su Jeong Wang, Sun-Young Oh, Han Uk Ryu
BACKGROUND: Cyclosporine A (CsA) is a widely used immunosuppressive agent that may provoke unexpected neurologic complications. The mechanism is unclear and variable intervals have been reported between CsA administration and onset of the related side effects. Here, we describe a case of delayed-onset CsA neurotoxicity presenting as opsoclonus-myoclonus syndrome (OMS). CASE DETAILS: A 37-year-old woman with a two-week period of opsoclonus and upper extremity myoclonus was admitted to our hospital...
September 14, 2017: Clinical Toxicology
https://www.readbyqxmd.com/read/28881516/-effectiveness-and-safety-of-rituximab-for-children-with-autoimmune-diseases-of-the-nervous-system
#13
Z Fu, X H Bao, Y Wu, J Zhou, Y H Zhang, Y Zhang, T Y Ji, Y Chen
Objective: To assess the effectiveness and safety of rituximab in Chinese children with autoimmune diseases of the nervous system. Method: An ambispective cohort study enrolled patients with refractory and(or) relapse autoimmune diseases of nervous system from June 2010 to June 2016 in Peking University First Hospital.These patients failed to respond to steroids and(or)intravenous immunoglobulin (IVIG) were treated with rituximab and seen for follow-up visits once every 3 months.The effectiveness was assessed by modified Rankin scale (mRs) and the annualized relapse rate...
September 2, 2017: Zhonghua Er Ke za Zhi. Chinese Journal of Pediatrics
https://www.readbyqxmd.com/read/28740856/opsoclonus-myoclonus-syndrome-during-rituximab-treatment-for-autoimmune-autonomic-ganglionopathy
#14
Oana M Dumitrascu, Andrew McKeon, Leslie Zuniga, Marie F Grill, Brent P Goodman
No abstract text is available yet for this article.
September 2017: Neurology® Neuroimmunology & Neuroinflammation
https://www.readbyqxmd.com/read/28710878/6-mercaptopurine-modifies-cerebrospinal-fluid-t-cell-abnormalities-in-paediatric-opsoclonus-myoclonus-as-steroid-sparer
#15
M R Pranzatelli, E D Tate, T J Allison
The purpose of this study was to evaluate the capacity of 6-mercaptopurine (6-MP), a known immunosuppressant, to normalize cerebrospinal fluid (CSF) lymphocyte frequencies in opsoclonus-myoclonus syndrome (OMS) and function as a steroid sparer. CSF and blood lymphocytes were immunophenotyped in 11 children with OMS (without CSF B cell expansion) using a comprehensive panel of cell surface adhesion, activation and maturation markers by flow cytometry, and referenced to 18 paediatric controls. Drug metabolites, lymphocyte counts and liver function tests were used clinically to monitoring therapeutic range and toxicity...
November 2017: Clinical and Experimental Immunology
https://www.readbyqxmd.com/read/28651977/dexamethasone-intravenous-immunoglobulin-and-rituximab-combination-immunotherapy-for-pediatric-opsoclonus-myoclonus-syndrome
#16
Michael R Pranzatelli, Elizabeth D Tate
BACKGROUND: Although pulse-dose dexamethasone is increasingly favored for treating pediatric opsoclonus-myoclonus syndrome (OMS), and multimodal immunotherapy is associated with improved clinical response, there have been no neuroimmunologic studies of dexamethasone-based multimodal disease-modifying therapy. METHODS: In this observational retrospective study, 19 children with OMS (with or without associated neuroblastoma) underwent multibiomarker evaluation for neuroinflammation...
August 2017: Pediatric Neurology
https://www.readbyqxmd.com/read/28549713/childhood-opsoclonus-myoclonus-syndrome-a-case-series-from-tunisia
#17
Nedia Ben Achour, Saloua Mrabet, Ibtihel Rebai, Ines Abid, Hanene Benrhouma, Hedia Klaa, Aida Rouissi, Ichraf Kraoua, Ilhem Ben Youssef Turki
INTRODUCTION: Opsoclonus myoclonus syndrome (OMS) is a rare immune-mediated disorder characterized by opsoclonus, myoclonus, ataxia and behavioral changes. The aim of our study was to investigate the epidemiology, clinical features, etiological aspects and outcome of OMS in Tunisian children. METHODS: We conducted a retrospective study over 11years (2005-2016) including all patients aged under 18years who were managed for newly diagnosed OMS in a tertiary care research centre for children with neurological symptoms...
May 23, 2017: Brain & Development
https://www.readbyqxmd.com/read/28479124/opsoclonus-myoclonus-syndrome-a-new-era-of-improved-prognosis
#18
Armine Galstyan, Colin Wilbur, Kathryn Selby, Juliette Hukin
BACKGROUND: Opsoclonus-myoclonus syndrome is an autoimmune neurological disorder characterized by opsoclonus, myoclonus, ataxia, and behavioral changes. Although long-term outcomes have historically been poor, including motor and cognitive disabilities, the advent of new and more aggressive immunotherapy regimens may be improving prognosis in opsoclonus-myoclonus syndrome. METHODS: We retrospectively reviewed the records of all children diagnosed with opsoclonus-myoclonus syndrome at BC Children's Hospital from 2000 to 2010...
July 2017: Pediatric Neurology
https://www.readbyqxmd.com/read/28460008/the-spectrum-and-burden-of-influenza-associated-neurological-disease-in-children-combined-encephalitis-and-influenza-sentinel-site-surveillance-from-australia-2013-2015
#19
P N Britton, C C Blyth, K Macartney, R C Dale, J Li-Kim-Moy, G Khandaker, N Crawford, H Marshall, J Clark, E Elliott, R Booy, A C Cheng, C A Jones
Background: There are few longitudinal studies of seasonal influenza associated neurological disease (IAND) and none from the Southern hemisphere. Methods: We extracted prospectively acquired Australian surveillance data from two studies nested within the Paediatric Active Enhanced Disease Surveillance (PAEDS) network: the Influenza Complications Alert Network (FluCAN) study and the Australian Childhood Encephalitis (ACE) study between 2013 and 2015. We described the clinical features and severity of IAND in children, including influenza associated encephalitis/encephalopathy (IAE)...
April 29, 2017: Clinical Infectious Diseases: An Official Publication of the Infectious Diseases Society of America
https://www.readbyqxmd.com/read/28432174/postintervention-acute-opsoclonus-myoclonus-syndrome
#20
Annelise Aquilina, Nicola Dingli, Josanne Aquilina
Opsoclonus myoclonus syndrome (OMS) is a rare, neurological condition affecting 1 in 10 000 000 people annually. Opsoclonus, defined as involuntary rapid, multivectorial oscillations of the eyes, together with ataxia and myoclonus are usually present. OMS may be paraneoplastic: often associated with occult neuroblastoma in childhood and with breast carcinoma or small cell lung carcinoma in adults. Other aetiologies include viral or toxic agents. The pathogenesis is thought to be immune mediated. A 37-year-old woman with previous inflammatory cranial mononeuropathies was admitted for elective dilatation and curettage (D&C)...
April 21, 2017: BMJ Case Reports
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