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opsoclonus myoclonus syndrome

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https://www.readbyqxmd.com/read/29773357/acute-transverse-myelitis-following-an-opsoclonus-myoclonus-syndrome-an-unusual-presentation
#1
Thomas Simon, Emmanuel Cheuret, Léa Fiedler, Catherine Mengelle, Eloïse Baudou, Kumaran Deiva
Opso-myoclonus syndrome (OMS) is a very rare and severe condition. Ataxia, opsoclonus, myoclonus and/or behavioral and sleeping disturbances define that autoimmune disorder syndrome which is paraneoplastic or triggered by an infection. Here, we report a 3 year-old immunocompetent boy who developed an atypical OMS which was later complicated by an acute transverse myelitis. Screening for neuroblastoma was negative and extensive infectious screening revealed an active HHV-6 infection confirmed by blood and cerebrospinal fluid PCR...
May 8, 2018: European Journal of Paediatric Neurology: EJPN
https://www.readbyqxmd.com/read/29742520/ipilimumab-nivolumab-related-opsoclonus-myoclonus-ataxia-syndrome-variant-in-a-patient-with-malignant-pleural-mesothelioma
#2
Bradley Maller, Edwin Peguero, Tawee Tanvetyanon
INTRODUCTION: Ipilimumab and nivolumab are immune-checkpoint inhibitors commonly used for melanoma. The combination is being investigated for its efficacy against several types of cancer, including malignant pleural mesothelioma. Although immune-related adverse events have been reported in patients receiving immune-checkpoint inhibitors, opsoclonus-myoclonus-ataxia syndrome has never been previously described. CASE PRESENTATION: We describe a 74-year-old male with malignant pleural mesothelioma who presented with opsoclonus and marked truncal ataxia ∼10 weeks following immunotherapy with ipilimumab and nivolumab...
May 8, 2018: Journal of Immunotherapy
https://www.readbyqxmd.com/read/29727049/multifactorial-analysis-of-opsoclonus-myoclonus-syndrome-etiology-tumor-vs-no-tumor-in-a-cohort-of-356-us-children
#3
Michael R Pranzatelli, Elizabeth D Tate, Nathan R McGee
BACKGROUND: Pediatric opsoclonus-myoclonus syndrome (OMS) presents a paradox of etiopathogenesis: A neuroblastic tumor (NB) is found in only one half of the cases, the others are ascribed to infections or designated as idiopathic. METHOD: From an IRB-approved observational study of 356 US children with OMS, secondary analysis of "etiology" and related factors was performed on a well-characterized cohort. The "Tumor" (n = 173) and "No Tumor" groups (n = 183), as defined radiologically, were compared according to multiple factors considered potentially differentiating...
May 4, 2018: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/29693780/autologous-stem-cell-transplantation-for-refractory-opsoclonus-myoclonus-ataxia-syndrome
#4
Donna L Johnston, Sean Murray, Meredith S Irwin, John Doyle, Tal Schechter
Opsoclonus, myoclonus, ataxia syndrome (OMA) is a severe neurologic disorder often associated with neuroblastoma. It is challenging to treat and can have long-term neurologic sequelae. Current recommended therapies include intravenous immunoglobulin, corticosteroids, rituximab, and chemotherapy (cyclophosphamide). We present two cases who were refractory to conventional therapy and underwent autologous stem cell transplantation (ASCT). One patient had complete resolution of symptoms following ASCT and the other patient had minimal change in symptoms with this therapy...
April 25, 2018: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/29685330/relation-of-intrathecal-oligoclonal-band-production-to-inflammatory-mediator-and-immunotherapy-response-in-208-children-with-oms
#5
Michael R Pranzatelli, Nathan R McGee, Elizabeth D Tate
In 208 children with opsoclonus-myoclonus syndrome (OMS), CSF IgG oligoclonal bands (OCB) and 22 immunomarkers in CSF and 21 in serum/blood were measured. In 36 untreated OMS, 58% were OCB(+), whereas 55% of treated OMS were OCB(-). OCB positivity or negativity did not alter concentrations or frequencies of immunomarkers. The phenotypes of OCB(+) and OCB(-) patients were not distinctive. CSF B cells were expanded in untreated OMS regardless of OCB positivity. These data reveal a much higher frequency of OCB positivity in untreated OMS than previously realized and a disconnect between intrathecal OCB and inflammatory mediator production...
April 12, 2018: Journal of Neuroimmunology
https://www.readbyqxmd.com/read/29561731/paraneoplastic-movement-disorders
#6
Karolina Popławska-Domaszewicz, Jolanta Florczak-Wyspiańska, Wojciech Kozubski, Sławomir Michalak
Paraneoplastic movement disorders are rare, autoimmune-mediated, nonmetastatic complications of malignant neoplasms. Common paraneoplastic movement disorders include paraneoplastic chorea, dystonia, cerebellar degeneration, different types of encephalitis, opsoclonus-myoclonus syndrome, stiff person syndrome, and neuromyotonia. Syndromes usually develop before tumor diagnosis, have subacute onset, and are associated with serum or cerebrospinal fluid antibodies. Two types of antibodies can be distinguished: antibodies against nuclear and cytoplasmic neuronal antigens (anti-Hu, anti-Ri, anti-Yo, anti-Ma, anti-CV2/CRMP5, anti-Gephrin, and anti-GABATRAP) and antibodies recently identified against cell surface and synaptic proteins (anti-NMDAR, anti-LGI1, and anti-Caspr2)...
March 21, 2018: Reviews in the Neurosciences
https://www.readbyqxmd.com/read/29555260/effect-of-low-dose-cyclophosphamide-acth-and-ivig-combination-immunotherapy-on-neuroinflammation-in-pediatric-onset-oms-a-retrospective-pilot-study
#7
Michael R Pranzatelli, Tyler J Allison, Elizabeth D Tate
INTRODUCTION: Flow cytometric cerebrospinal fluid (CSF) lymphocyte subset analysis has improved the diagnosis of neuroinflammation and identified multiple markers of inflammation in opsoclonus-myoclonus syndrome (OMS). The aim of this exploratory, retrospective study was to analyze the effect of immunotherapy on these markers to determine which agents are disease modifying. METHODS: Cross-sectional immunological observations were made in an IRB-approved case-control study, and patients were treated empirically...
March 5, 2018: European Journal of Paediatric Neurology: EJPN
https://www.readbyqxmd.com/read/29522119/opsoclonus-myoclonus-syndrome-additional-clinical-considerations
#8
David Dongkyung Kim, Adrian Budhram
No abstract text is available yet for this article.
April 1, 2018: JAMA Otolaryngology—Head & Neck Surgery
https://www.readbyqxmd.com/read/29522060/opsoclonus-myoclonus-syndrome-reply
#9
Shuo-Yen Hsu, Yi-Ho Young
No abstract text is available yet for this article.
April 1, 2018: JAMA Otolaryngology—Head & Neck Surgery
https://www.readbyqxmd.com/read/29485697/cerebrospinal-fluid-%C3%AE-%C3%AE-t-cell-frequency-is-age-related-a-case-control-study-of-435-children-with-inflammatory-and-non-inflammatory-neurological-disorders
#10
M R Pranzatelli, T J Allison, N R McGee, E D Tate
Studies of cerebrospinal fluid (CSF) γδ T cells in children are limited, due especially to the lack of control data. In adults, gamma/delta T cells (TCR-γδ) residing in the intrathecal space are sometimes involved in neuroinflammation. To evaluate the possible role of γδ T cells in paediatric neuroinflammation, we immunophenotyped cerebrospinal fluid (CSF) and blood lymphocytes using flow cytometry in a case-control study of 100 children with non-inflammatory neurological disorders (NIND), 312 with opsoclonus-myoclonus (OMS) and 23 with other inflammatory neurological disorders (OIND)...
February 27, 2018: Clinical and Experimental Immunology
https://www.readbyqxmd.com/read/29406902/autoimmune-and-paraneoplastic-movement-disorders-an-update
#11
REVIEW
José Fidel Baizabal-Carvallo, Joseph Jankovic
Movement disorders (MDs) are common in patients with autoimmune disorders affecting the central and peripheral nervous system. They may be observed in autoimmune disorders triggered by an infectious agent, such as streptococcus in Sydenham's chorea, or in basal ganglia encephalitis with antibodies against the dopamine-D2 receptors. In these patients chorea or dystonia are usually the most prominent hyperkinetic MDs. MDs are also observed in patients with diffuse or limbic encephalitis with antibodies directed against neuronal cell-surface antigens...
February 15, 2018: Journal of the Neurological Sciences
https://www.readbyqxmd.com/read/29376112/intravenous-immunoglobulin-with-prednisone-and-risk-adapted-chemotherapy-for-children-with-opsoclonus-myoclonus-ataxia-syndrome-associated-with-neuroblastoma-anbl00p3-a-randomised-open-label-phase-3-trial
#12
Pedro A de Alarcon, Katherine K Matthay, Wendy B London, Arlene Naranjo, Sheena C Tenney, Jessica A Panzer, Michael D Hogarty, Julie R Park, John M Maris, Susan L Cohn
Purpose: No previous clinical trial has been conducted for patients with neuroblastoma associated opsoclonus myoclonus ataxia syndrome (OMA), and current treatment is based on case reports. To evaluate the OMA response to prednisone and risk-adapted chemotherapy and determine if the addition of intravenous gammaglobulin (IVIG) further improves response, the Children's Oncology Group designed a randomized therapeutic trial. Patient and Methods: Eligible subjects were randomized to receive twelve cycles of IVIG (IVIG+) or no IVIG (NO-IVIG) in addition to prednisone and neuroblastoma risk-adapted chemotherapy...
January 2018: Lancet Child & Adolescent Health
https://www.readbyqxmd.com/read/29289523/principles-and-approaches-to-the-treatment-of-immune-mediated-movement-disorders
#13
REVIEW
Shekeeb S Mohammad, Russell C Dale
Immune mediated movement disorders include movement disorders in the context of autoimmune encephalitis such as anti-NMDAR encephalitis, post-infectious autoimmune movement disorders such as Sydenham chorea, paraneoplastic autoimmune movement disorders such as opsoclonus myoclonus ataxia syndrome, and infection triggered conditions such as paediatric acute neuropsychiatric syndrome. This review focuses on the approach to treatment of immune mediated movement disorders, which requires an understanding of the immunopathogenesis, whether the disease is destructive or 'altering', and the natural history of disease...
March 2018: European Journal of Paediatric Neurology: EJPN
https://www.readbyqxmd.com/read/29243132/hiv-associated-opsoclonus-myoclonus-ataxia-syndrome-early-infection-immune-reconstitution-syndrome-or-secondary-to-other-diseases-case-report-and-literature-review
#14
Bruno F Guedes, Márcio A A Vieira Filho, Clarice Listik, Rafael B Carra, Cristiane B Pereira, Emanuelle R da Silva, Hélio R Gomes, José E Vidal
Opsoclonus-myoclonus-ataxia (OMA) syndrome is a debilitating autoimmune neurological disorder. Post-infectious opsoclonus-myoclonus-ataxia syndrome has been described with varying bacterial, spirochetal, and viral infections including several patients with HIV. However, specific immunopathological mechanisms that may lead to opsoclonus-myoclonus in HIV-positive patients are unknown.We report a case of HIV-associated opsoclonus-myoclonus and early HIV infection. A review of published literature shows opsoclonus-myoclonus can occur during early infection, in immune reconstitution syndrome or in association with other infections, especially tuberculosis...
February 2018: Journal of Neurovirology
https://www.readbyqxmd.com/read/29169865/corrigendum-to-dexamethasone-intravenous-immunoglobulin-and-rituximab-combination-immunotherapy-for-pediatric-opsoclonus-myoclonus-syndrome-pediatric-neurology-73-2017-48-56
#15
Michael R Pranzatelli, Elizabeth D Tate
No abstract text is available yet for this article.
January 2018: Pediatric Neurology
https://www.readbyqxmd.com/read/29135842/genomic-profiles-of-neuroblastoma-associated-with-opsoclonus-myoclonus-syndrome
#16
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...
March 2018: 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
#17
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...
December 2017: Clinical Neurology and Neurosurgery
https://www.readbyqxmd.com/read/29103425/cancer-and-autoimmunity-paraneoplastic-neurological-disorders-associated-with-neuroblastic-tumors
#18
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
#19
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...
November 2017: Parkinsonism & related Disorders
https://www.readbyqxmd.com/read/29058581/opsoclonus-myoclonus-syndrome-in-a-patient-with-japanese-encephalitis-a-case-report
#20
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
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