RESUMO
Opsoclonus-myoclonus syndrome is a combination of involuntary, arrhythmic, conjugate saccadic eye movements with myoclonus. The most common cause in adults is paraneoplastic encephalitis. Rarer causes include infections such as scrub typhus, and toxins such as organophosphates and cocaine. Organophosphates are one of the common poisonings in tropical countries such as India, causing both central and peripheral nervous system manifestations. We describe a middle-aged male farmer with unexplained altered consciousness and respiratory depression. After 2 days, he developed opsoclonus-myoclonus, and then bronchorrhoea and bradycardia, raising suspicion of organophosphate poisoning. After we had identified a very low serum cholinesterase concentration, he disclosed having consumed organophosphates.
Assuntos
Cocaína , Encefalite , Síndrome de Opsoclonia-Mioclonia , Intoxicação por Organofosfatos , Adulto , Pessoa de Meia-Idade , Humanos , Masculino , Síndrome de Opsoclonia-Mioclonia/induzido quimicamente , Intoxicação por Organofosfatos/complicações , Intoxicação por Organofosfatos/diagnóstico , ÍndiaRESUMO
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. After three asymptomatic years, he relapsed. Upon reintroducing TPE and rituximab plus oral prednisolone, the patient rapidly achieved a second complete remission. This case offers proof-of-principle for the potential efficacy of TPE for neuroblastoma-associated OMAS.
Assuntos
Protocolos de Quimioterapia Combinada Antineoplásica/efeitos adversos , Neuroblastoma/tratamento farmacológico , Síndrome de Opsoclonia-Mioclonia/terapia , Síndromes Paraneoplásicas Oculares/tratamento farmacológico , Troca Plasmática , Protocolos de Quimioterapia Combinada Antineoplásica/administração & dosagem , Humanos , Lactente , Masculino , Síndrome de Opsoclonia-Mioclonia/induzido quimicamenteRESUMO
Opsoclonus-myoclonus syndrome (OMS) is characterized by abnormal eye and systemic involuntary movements, as well as cerebellar ataxia. Some sedatives and anesthetics worsen movements associated with OMS, while there is no known report of a negative effect of atropine. We report on sedation in two patients with OMS. Involuntary movements were transiently worsened after using atropine with midazolam or thiamylal in both, but were not seen when atropine was not used. We speculated that atropine has the potential to exacerbate involuntary movements in OMS due to vulnerability to this agent via unknown mechanisms.
Assuntos
Adjuvantes Anestésicos/efeitos adversos , Atropina/efeitos adversos , Síndrome de Opsoclonia-Mioclonia/induzido quimicamente , Anticonvulsivantes/uso terapêutico , Pré-Escolar , Clonazepam/uso terapêutico , Sedação Profunda/efeitos adversos , Sedação Profunda/métodos , Quimioterapia Combinada , Feminino , Glucocorticoides/uso terapêutico , Humanos , Lactente , Imageamento por Ressonância Magnética , Síndrome de Opsoclonia-Mioclonia/diagnóstico por imagem , Síndrome de Opsoclonia-Mioclonia/fisiopatologia , Prednisolona/uso terapêutico , Estudos Retrospectivos , Tomografia Computadorizada por Raios XRESUMO
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. The patient had been taking CsA for 17 years after receiving a kidney transplant. Further evaluation did not reveal any other abnormalities. Seven days after switching from CsA to tacrolimus, in the absence of additional immune-modulating therapy, her neurologic symptoms improved considerably. CONCLUSION: This is the case of delayed, long-term complications of CsA presenting as OMS. Symptoms resolved by substituting CsA with another immunomodulating drug. The etiology of the neurologic complications may involve paradoxically-enhanced delayed-type hypersensitivity.
Assuntos
Ciclosporina/efeitos adversos , Imunossupressores/efeitos adversos , Síndrome de Opsoclonia-Mioclonia/induzido quimicamente , Adulto , Ciclosporina/uso terapêutico , Feminino , Humanos , Imunossupressores/uso terapêutico , Transplante de Rim/efeitos adversosRESUMO
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 â¼10 weeks following immunotherapy with ipilimumab and nivolumab. No myoclonus was present. Oligoclonal bands were detected in cerebrospinal fluid. Treatment with methylprednisolone and intravenous immunoglobulin along with clonazepam and valproic acid resulted in a rapid clinical improvement. A follow-up visit 2 months afterward showed a resolution of opsoclonus and he was able to walk with cane. CONCLUSIONS: A variant of opsoclonus-myoclonus-ataxia syndrome may occur following treatment with ipilimumab and nivolumab.
Assuntos
Antineoplásicos Imunológicos/efeitos adversos , Ataxia/induzido quimicamente , Ipilimumab/efeitos adversos , Nivolumabe/efeitos adversos , Síndrome de Opsoclonia-Mioclonia/induzido quimicamente , Idoso , Ataxia/tratamento farmacológico , Clonazepam/uso terapêutico , Humanos , Imunoglobulinas/uso terapêutico , Imunossupressores/uso terapêutico , Neoplasias Pulmonares/tratamento farmacológico , Masculino , Mesotelioma/tratamento farmacológico , Mesotelioma Maligno , Metilprednisolona/uso terapêutico , Síndrome de Opsoclonia-Mioclonia/tratamento farmacológico , Neoplasias Pleurais/tratamento farmacológico , Ácido Valproico/uso terapêuticoAssuntos
Antibacterianos/efeitos adversos , Cefepima/efeitos adversos , Síndrome de Opsoclonia-Mioclonia/induzido quimicamente , Idoso , Anticonvulsivantes/uso terapêutico , Eletroencefalografia , Feminino , Cefaleia/tratamento farmacológico , Humanos , Levetiracetam , Síndrome de Opsoclonia-Mioclonia/tratamento farmacológicoRESUMO
We report the case of a 38-year-old woman with AIDS who developed opsoclonus-myoclonus-ataxia syndrome during a period different from other cases reported in literature. Opsoclonus-myoclonus-ataxia syndrome had already been reported as the initial neurological presentation of AIDS, as well as at the time of HIV-seroconversion and immune reconstitution syndrome. Our case is unique since the patient had an elevated CD4 count and negative viral load in the period when the opsoclonus-myoclonus-ataxia syndrome occurred.
Assuntos
Terapia Antirretroviral de Alta Atividade/efeitos adversos , Ataxia/induzido quimicamente , Infecções por HIV/complicações , Síndrome de Opsoclonia-Mioclonia/induzido quimicamente , Transtornos Parkinsonianos/induzido quimicamente , Adulto , Ataxia/patologia , Encéfalo/patologia , Feminino , Infecções por HIV/tratamento farmacológico , Humanos , Síndrome Inflamatória da Reconstituição Imune/complicações , Síndrome Inflamatória da Reconstituição Imune/diagnóstico , Imageamento por Ressonância Magnética , Síndrome de Opsoclonia-Mioclonia/diagnóstico , Transtornos Parkinsonianos/diagnóstico , Carga ViralAssuntos
Antidepressivos de Segunda Geração/efeitos adversos , Ataxia/induzido quimicamente , Síndrome de Opsoclonia-Mioclonia/induzido quimicamente , Cloridrato de Venlafaxina/efeitos adversos , Adulto , Ataxia/complicações , Depressão/complicações , Depressão/tratamento farmacológico , Feminino , Humanos , Síndrome de Opsoclonia-Mioclonia/complicações , Transtornos de Estresse Pós-Traumáticos/complicações , Transtornos de Estresse Pós-Traumáticos/tratamento farmacológicoRESUMO
É relatado aqui o caso de uma mulher de 38 anos com AIDS que desenvolveu a síndrome de opsoclonia-mioclonia-ataxia em um período diferente dos outros casos já relatados na literatura. A síndrome de opsoclonia-mioclonia-ataxia já tinha sido relatada como manifestação inicial de AIDS, assim como no momento da soroconversão de HIV e na síndrome de reconstituição imune. Este caso é único, uma vez que a paciente tinha contagem elevada de CD4 e carga viral negativa no momento em que a síndrome de opsoclonia-mioclonia-ataxia ocorreu.
We report the case of a 38-year-old woman with AIDS who developed opsoclonus-myoclonus-ataxia syndrome during a period different from other cases reported in literature. Opsoclonus-myoclonus-ataxia syndrome had already been reported as the initial neurological presentation of AIDS, as well as at the time of HIV-seroconversion and immune reconstitution syndrome. Our case is unique since the patient had an elevated CD4 count and negative viral load in the period when the opsoclonus-myoclonus-ataxia syndrome occurred.