Brain volumetric analysis and cortical thickness in adults with saccadic intrusions (ocular flutter or opsoclonus-myoclonus syndrome)

Ibáñez-Juliá, María-José; Pappa, Evangelia; Gaymard, Bertrand; Leclercq, Delphine; Hautefort, Charlotte; Tilikete, Caroline; Delattre, Jean‐Yves; Hoang-Xuân, Khê; Psimaras, Dimitri; Alentorn, Agustí

doi:10.1016/j.clineuro.2017.10.028

Cited by 4 publications

(3 citation statements)

References 27 publications

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“…8 Furthermore, reductions in cortical thickness (motor and visual cortices) and subcortical gray matter have been documented at chronic stages of the disease. 10,14 Our findings, in which the cerebellar gray matter volume in children with opsoclonus-myoclonus ataxia syndrome diverges from healthy children several years after disease onset, suggest that despite the use of aggressive therapies, a degree of injury may be sustained, which ultimately affects volume changes through time. Our data demonstrate differences between the expected inverted U-shaped trajectory of cerebellar gray matter growth in healthy children and children with opsoclonus-myoclonus ataxia syndrome, suggesting deviations in both the normal growth and pruning processes of the cerebellar gray matter.…”

Section: Discussionmentioning

confidence: 80%

Brain Volumes in Opsoclonus-Myoclonus Ataxia Syndrome: A Longitudinal Study

Almudhry,

Wagner,

Longoni

et al. 2024

J Child Neurol

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Introduction Little is known about the longitudinal trajectory of brain growth in children with opsoclonus-myoclonus ataxia syndrome. We performed a longitudinal evaluation of brain volumes in pediatric opsoclonus-myoclonus ataxia syndrome patients compared with age- and sex-matched healthy children. Patients and Methods This longitudinal case-control study included brain magnetic resonance imaging (MRI) scans from consecutive pediatric opsoclonus-myoclonus ataxia syndrome patients (2009-2020) and age- and sex-matched healthy control children. FreeSurfer analysis provided automatic volumetry of the brain. Paired t tests were performed on the curvature of growth trajectories, with Bonferroni correction. Results A total of 14 opsoclonus-myoclonus ataxia syndrome patients (12 female) and 474 healthy control children (406 female) were included. Curvature of the growth trajectories of the cerebral white and gray matter, cerebellar white and gray matter, and brainstem differed significantly between opsoclonus-myoclonus ataxia syndrome patients and healthy control children (cerebral white matter, P = .01; cerebral gray matter, P = .01; cerebellar white matter, P < .001; cerebellar gray matter, P = .049; brainstem, P < .01). Discussion/Conclusion We found abnormal brain maturation in the supratentorial brain, brainstem, and cerebellum in children with opsoclonus-myoclonus ataxia syndrome.

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Section: Discussionmentioning

confidence: 80%

Brain Volumes in Opsoclonus-Myoclonus Ataxia Syndrome: A Longitudinal Study

Almudhry,

Wagner,

Longoni

et al. 2024

J Child Neurol

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“…Details about imaging findings on follow-up brain MRI scans performed weeks or months after the acute illness were not available. However, a recent small study comparing brain volumetry of healthy controls and patients with previous ocular flutter or opsoclonus showed a decrease of the cerebellar cortex volume with preservation of the cerebellar white matter volume and a subsequent decrease in both cortical and subcortical gray matter of the cerebral hemispheres despite the fact that most of the patients made a complete recovery ( 47 ). EEG findings were reported in few patients and showed a non-specific diffuse slow rhythm.…”

Section: Discussionmentioning

confidence: 99%

Opsoclonus-Myoclonus Syndrome Associated With West-Nile Virus Infection: Case Report and Review of the Literature

et al. 2018

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Opsoclonus-myoclonus syndrome (OMS) is a very rare condition with different autoimmune, infectious and paraneoplastic aetiologies or in most cases idiopathic. We report the case of a 75-year-old woman who was admitted in our department in early fall for altered mental status, opsoclonus, multifocal myoclonus, truncal titubation and generalized tremor, preceded by a 5 day prodrome consisting of malaise, nausea, fever and vomiting. Brain computed tomography and MRI scans showed no significant abnormalities and cerebrospinal fluid changes consisted of mildly increased protein content and number of white cells. Work-up for paraneoplastic and autoimmune causes of OMS was negative but serologic tests identified positive IgM and IgG antibodies against West Nile virus (WNV). The patient was treated with Dexamethasone and Clonazepam with progressive improvement of mental status, myoclonus, opsoclonus and associated neurologic signs. Six months after the acute illness she had complete recovery. To our knowledge this is the 14th case of WNV associated OMS reported in the literature so far. We briefly describe the clinical course of the other reported cases together with the different treatment strategies that have been employed.

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“…Opsoclonus and ocular flutter are both unwanted saccades that disrupt steady fixation. Opsoclonus differs from ocular flutter in that the saccade consists of several planes, including horizontal, vertical, and torsional components, rather than just the horizontal plane (14,15) . Opsoclonus is a saccade, as opposed to nystagmus, which is a rhythmic and slow oscillation that takes the eyes away from the target (16,17) .…”

Section: Presentation and Diagnosismentioning

confidence: 99%

Adult-onset idiopathic opsoclonus-myoclonus syndrome

Zhang

Yan

Song

et al. 2023

ABO

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Opsoclonus-myoclonus syndrome is extremely uncommon in adults with an autoimmune pathophysiology. Because of the rarity of the syndrome, international recognition of opsoclonus-myoclonus-ataxia syndrome needs to be improved urgently. Therefore, the goal of this study was to raise the awareness of the opsoclonus-myoclonus-ataxia syndrome and help doctors in better diagnosing and using immunotherapy. Methods: We present a case study of an adult-onset case of idiopathic opsoclonus-myoclonus syndrome characterized by spontaneous arrhythmic multidirectional conjugate eye movements, myoclonus, ataxia, sleep disorders, and intense fear. Additionally, we conduct a literature search and summarize the pathophysiology, clinical presentation, diagnosis, and treatment of opsoclonus-myoclonus-ataxia syndrome. Results: Immunotherapies successfully treated the patient's opsoclonus, myoclonus, and ataxia. Further, the article also includes an update summary of the opsoclonus--myoclonus-ataxia syndrome. Conclusion: The prevalence of residual sequela in adults with opsoclonus-myoclonus-ataxia syndrome is low. Early diagnosis and treatment may result in a better prognosis. Furthermore, combined immunotherapy is expected to reduce the incidence of refractory and reoccurring opsoclonus-myoclonus-ataxia syndrome.

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Brain volumetric analysis and cortical thickness in adults with saccadic intrusions (ocular flutter or opsoclonus-myoclonus syndrome)

Cited by 4 publications

References 27 publications

Brain Volumes in Opsoclonus-Myoclonus Ataxia Syndrome: A Longitudinal Study

Brain Volumes in Opsoclonus-Myoclonus Ataxia Syndrome: A Longitudinal Study

Opsoclonus-Myoclonus Syndrome Associated With West-Nile Virus Infection: Case Report and Review of the Literature

Adult-onset idiopathic opsoclonus-myoclonus syndrome

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