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Opsoclonus myoclonus

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https://www.readbyqxmd.com/read/30221603/immune-mediated-cerebellar-ataxias-practical-guidelines-and-therapeutic-challenges
#1
Hiroshi Mitoma, Mario Manto, Christiane S Hampe
Immune-mediated cerebellar ataxias (IMCAs), a clinical entity reported for the first time in the 1980s, include gluten ataxia (GA), paraneoplastic cerebellar degenerations (PCDs), anti-glutamate decarboxylase 65 (GAD) antibody-associated cerebellar ataxia, post-infectious cerebellitis, and opsoclonus myoclonus syndrome (OMS). These IMCAs share common features with regard to therapeutic approaches. When certain factors trigger immune processes, elimination of the antigen(s) becomes a priority: e.g., gluten-free diet in GA and surgical excision of the primary tumor in PCDs...
September 16, 2018: Current Neuropharmacology
https://www.readbyqxmd.com/read/30197220/evaluation-of-responsiveness-to-reduced-dose-rituximab-in-corticotropin-intravenous-immunoglobulin-rituximab-combination-immunotherapy-for-opsoclonus-myoclonus-syndrome
#2
Michael R Pranzatelli, Elizabeth D Tate, Nathan R McGee, Craig A MacArthur
BACKGROUND: Rituximab (anti-CD20) has been used as B-cell-targeted intervention to treat opsoclonus-myoclonus syndrome. Due to isolated reports of chronic hypogammaglobulinemia and B lymphopenia following rituximab in several disorders, and rapid B-cell depletion after a few doses, we reduced the dosage 20% in our clinical practice. METHODS: In this Institutional Review Board-approved retrospective study, 32 children with opsoclonus-myoclonus syndrome and cerebrospinal fluid B-cell expansion had received front-loaded adrenocorticotropic hormone, intravenous immunoglobulin, and rituximab combination immunotherapy for de novo opsoclonus-myoclonus syndrome...
May 18, 2018: Pediatric Neurology
https://www.readbyqxmd.com/read/30169194/enigmatic-entities-opsoclonus-myoclonus-ataxia-syndrome-linked-to-neuroblastoma
#3
Brian H Kushner, Yasmin Khakoo
No abstract text is available yet for this article.
January 2018: Lancet Child & Adolescent Health
https://www.readbyqxmd.com/read/30141794/-psychological-development-of-children-with-opsoclonus-myoclonus-syndrome-and-its-correlation-with-age-and-disease-onset
#4
N S Burlakova, Yu V Mikadze, Yu N Fedorova, E S Ilyina, R Ts Bembeeva, N N Zavadenko
Opsoclonus-myoclonus syndrome (OMS) is a rare neurological disorder with unknown etiology and pathogenesis. There is not enough knowledge about psychological development of children with OMS. AIM: To study an influence of the time of disease onset and children's age and type of disease course on psychological development of children with OMS. MATERIAL AND METHODS: The study included 26 children with OMS of age between 1 year 7 months and 13 years, 10 boys and 16 girls...
2018: Zhurnal Nevrologii i Psikhiatrii Imeni S.S. Korsakova
https://www.readbyqxmd.com/read/30124121/anti-ri-associated-paraneoplastic-neurological-syndrome-initial-symptom-of-breast-cancer-with-her2-overexpression-and-treatment-by-dual-her2-blockade
#5
Omer Fatih Olmez, Oguzcan Kinikoglu, Nesrin Helvacı Yilmaz, Ahmet Bilici, Erdem Cubukcu, Mehmet Seker, Tansel Cakir, Ozcan Yildiz, Jamshid Hamdard
Paraneoplastic neurological syndrome is associated with anti-Ri antibodies, which are typically present with opsoclonus-myoclonus-ataxia. Human epidermal growth factor receptor 2 (HER2) overexpression is present in 15%-25% of breast cancer and is associated with poor prognosis. There are a few reports of paraneoplastic neurological syndrome associated with HER2-positive breast cancer in the literature, of which most are anti-Yo-associated paraneoplastic neurological syndrome. We present herein the case of a female patient with HER2-positive breast cancer who had atypical anti-Ri antibody associated with opsoclonus-myoclonus paraneoplastic neurological syndrome...
August 19, 2018: Journal of Oncology Pharmacy Practice
https://www.readbyqxmd.com/read/30076990/opsoclonus-myoclonus-ataxia-syndrome-associated-with-chikungunya-and-dengue-virus-co-infection
#6
Mateus Santana do Rosário, Marta Giovanetti, Pedro Antonio Pereira de Jesus, Daniel Santana Farias, Nuno R Faria, Clayton Pereira S de Lima, Sandro Patroca da Silva, Marcio Roberto Nunes, Luiz Carlos Junior Alcantara, Isadora Cristina de Siqueira
Opsoclonus-myoclonus-ataxia syndrome (OMAS) is a rare neurological disorder characterized by irregular multidirectional eye movements, myoclonus, cerebellar ataxia, sleep disturbances, and cognitive dysfunction. Although most commonly related to paraneoplastic syndrome, this condition has occasionally been described following infectious illnesses. This article reports the first case of OMAS in association with chikungunya and dengue virus co-infection. The genetic analysis identified chikungunya virus of East/Central/South African genotype and dengue serotype 4 virus of genotype II...
August 1, 2018: International Journal of Infectious Diseases: IJID
https://www.readbyqxmd.com/read/30057463/prognostic-factors-in-anti-neuronal-antibody-positive-patients
#7
Çağla Aydin, Şenay Yıldız Çelik, Sema İçöz, Canan Ulusoy, Tuncay Gündüz, Gülşen Akman Demir, Murat Kürtüncü, Erdem Tüzün
Introduction: Anti-neuronal antibodies (ANA) are found in paraneoplastic neurological syndrome and autoimmune encephalitis patients. Our aim was to analyze prognostic factors related with ANA seropositivity. Methods: Twenty-seven consecutive ANA seropositive patients were included in the study. ANA were detected by immunofluorescent staining, immunoblot and cell-based assay methods. All patients were followed with a standard treatment protocol. Clinical syndromes, tumor types, modified Rankin scores, cranial MRI and oligoclonal band (OCB) findings were recorded...
2018: Noro Psikiyatri Arsivi
https://www.readbyqxmd.com/read/30045961/glutamate-receptor-%C3%AE-2-serum-antibodies-in-pediatric-opsoclonus-myoclonus-ataxia-syndrome
#8
Georgina Berridge, David A Menassa, Teresa Moloney, Patrick J Waters, Imogen Welding, Selina Thomsen, Sameer Zuberi, Roman Fischer, A Radu Aricescu, Michael Pike, Russell C Dale, Benedikt Kessler, Angela Vincent, Ming Lim, Sarosh R Irani, Bethan Lang
OBJECTIVE: To identify neuronal surface antibodies in opsoclonus myoclonus ataxia syndrome (OMAS) using contemporary antigen discovery methodology. METHODS: OMAS patient serum immunoglobulin G immunohistochemistry using age-equivalent rat cerebellar tissue was followed by immunoprecipitation, gel electrophoresis, and mass spectrometry. Data are available via ProteomeXchange (identifier PXD009578). This generated a list of potential neuronal surface cerebellar autoantigens...
August 21, 2018: Neurology
https://www.readbyqxmd.com/read/29891101/-paraneoplastic-syndromes-in-ophthalmology-french-version-of-the-article
#9
REVIEW
A Bussat, S Langner-Lemercier, A Salmon, F Mouriaux
Paraneoplastic syndromes involving the visual system are a heterogeneous group of disorders occurring in the setting of systemic malignancy. Although these syndromes are rare, the clinical manifestations can herald an unsuspected, underlying malignancy. The associated antibodies and histopathology of each syndrome are presented to help in the understanding of these autoimmune phenomena. Outlined in this review article are diagnostic features useful in differentiating cancer-associated retinopathy, melanoma-associated retinopathy, paraneoplastic polymorphous vitelliform maculopathy, bilateral diffuse uveal melanocytic proliferation and paraneoplastic neurologic syndromes such as optic neuropathy, opsoclonus-myoclonus, Lambert-Eaton myasthenia and paraneoplastic cerebellar degeneration...
June 2018: Journal Français D'ophtalmologie
https://www.readbyqxmd.com/read/29891069/immune-mediated-ataxias
#10
Bastien Joubert, Kevin Rostásy, Jérôme Honnorat
Immune-mediated cerebellar ataxia (CA) comprises a group of rare diseases that are still incompletely described, and are probably underdiagnosed. Both acute and progressive progressions are possible. Different syndromes have been identified, including CA associated with anti-GAD antibodies, the cerebellar type of Hashimoto encephalopathy, primary autoimmune CA, gluten ataxia, opsoclonus-myoclonus syndrome, and paraneoplastic cerebellar degenerations. Most of these syndromes are associated with autoantibodies targeting neuronal antigens...
2018: Handbook of Clinical Neurology
https://www.readbyqxmd.com/read/29784239/paraneoplastic-syndromes-in-ophthalmology
#11
REVIEW
A Bussat, S Langner-Lemercier, A Salmon, F Mouriaux
Paraneoplastic syndromes involving the visual system are a heterogeneous group of disorders occurring in the setting of systemic malignancy. They are unrelated to local tumor invasion or metastasis and unexplained by nutritional, metabolic, infectious or iatrogenic causes. The presence of antibodies may aid in the diagnosis of a paraneoplastic syndrome, although this is not an absolute requirement. Chorioretinal involvement is seen in CAR (cancer-associated retinopathy) syndrome, MAR (melanoma-associated retinopathy) syndrome, paraneoplastic vitelliform maculopathy, and bilateral diffuse uveal melanocytic proliferation...
May 2018: Journal Français D'ophtalmologie
https://www.readbyqxmd.com/read/29773357/acute-transverse-myelitis-following-an-opsoclonus-myoclonus-syndrome-an-unusual-presentation
#12
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...
September 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
#13
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
#14
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...
August 2018: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/29693780/autologous-stem-cell-transplantation-for-refractory-opsoclonus-myoclonus-ataxia-syndrome
#15
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...
August 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
#16
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...
August 15, 2018: Journal of Neuroimmunology
https://www.readbyqxmd.com/read/29561731/paraneoplastic-movement-disorders
#17
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
#18
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...
July 2018: European Journal of Paediatric Neurology: EJPN
https://www.readbyqxmd.com/read/29522119/opsoclonus-myoclonus-syndrome-additional-clinical-considerations
#19
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
#20
Shuo-Yen Hsu, Yi-Ho Young
No abstract text is available yet for this article.
April 1, 2018: JAMA Otolaryngology—Head & Neck Surgery
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