Ibáñez-Juliá María-José, Pappa Evangelia, Gaymard Bertrand, Leclercq Delphine, Hautefort Charlotte, Tilikete Caroline, Delattre Jean-Yves, Hoang-Xuan Khê, Psimaras Dimitri, Alentorn Agusti
Department of Neurology 2, Mazarin, Groupe Hospitalier Pitié-Salpêtrière, Assistance Publique Hôpitaux de Paris (AP-HP), 75013, Paris, France; Sorbonne Universités, Université Pierre et Marie Curie, Université Paris VI, 75013, Paris, France; Institut du Cerveau et de la Moelle Epinière, INSERM U1127 and Centre National de la Recherche Scientifique, Unité de Recherche Mixte, 7225, Paris, France.
Deparment of Clinical Neurophysiology, Groupe Hospitalier Pitié-Salpêtrière, Assistance Publique Hôpitaux de Paris (AP-HP), 75013, Paris, France.
Clin Neurol Neurosurg. 2017 Dec;163:167-172. doi: 10.1016/j.clineuro.2017.10.028. Epub 2017 Oct 31.
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.
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.
Patients with OF/Opsoclonus showed reduced cerebellum cortical volume with a subsequent diminution in total cerebellar volume. White mater cerebellum volume was not modified. In addition, we have also identified a significant supratentorial gray matter volume decrease in OF/Opsoclonus patients, involving both the cortical and the subcortical gray matter.
OF/Opsoclonus in adults may be associated with cortical and subcortical gray matter atrophy, as well as decreased cerebellar cortical volume. Further larger prospective studies are necessary to confirm these results.
眼球扑动(OF)和眼阵挛被认为是具有相似发病机制的连续体。由于该疾病在成年人群中罕见,对于其疾病慢性期的脑形态学变化知之甚少。
分析了6例有OF/眼阵挛既往史的成年人的磁共振成像以及12例健康患者(按年龄和性别配对)的磁共振成像,以便使用Freesurfer软件确定这种罕见疾病的长期皮质厚度模式。
OF/眼阵挛患者的小脑皮质体积减小,随后小脑总体积减小。小脑白质体积未改变。此外,我们还发现OF/眼阵挛患者幕上灰质体积显著减少,包括皮质和皮质下灰质。
成年人的OF/眼阵挛可能与皮质和皮质下灰质萎缩以及小脑皮质体积减小有关。需要进一步开展更大规模的前瞻性研究来证实这些结果。