Kharytaniuk N, Hikmat D, Ozkan H, Lim E A, Bamiou D E, Cowley P, Jäger H R, Werring D J
UCL Ear Institute, University College London, 332-336 Gray's Inn Road, London, WC1X 8EE, UK.
National Institute for Health and Care Research University College London Hospitals Biomedical Research Centre (Deafness and Hearing Problems Theme), London, UK.
J Neurol. 2025 Mar 4;272(3):242. doi: 10.1007/s00415-025-12965-w.
Classical infratentorial superficial siderosis (iSS) is considered a rare but potentially disabling neurological disorder. It results from slow chronic blood extravasation into the cerebrospinal fluid and deposition of the iron degradation product hemosiderin within the subpial layers of the central nervous system. Susceptibility-weighted (SW) magnetic resonance imaging (MRI) is the reference diagnostic modality. Few studies have described the population prevalence of cerebellar or infratentorial siderosis, and there are none from the UK population. The aim of this cross-sectional observational study was to estimate the prevalence of iSS in the UK Biobank sample using pre-defined radiological criteria.
We reviewed SW MRIs of participants from the UK Biobank, looking for the radiological features of classical iSS: involvement of infratentorial structures (superior vermis, cerebellar folia, brainstem, or craniocervical junction). We calculated the point prevalence as the number of identified cases per total number of cases reviewed and 95% confidence intervals (CI) using Wilson's Score formula.
Of 10,305 SW MRIs reviewed, five cases with radiological features of iSS were identified demonstrating cerebellar/superior vermis involvement. The estimated prevalence of iSS was calculated as 48.5 (95%CI 20.7-113.5) cases per 100,000 population.
This is the first study to estimate iSS prevalence in the UK population. The prevalence of iSS is higher than that reported for other rare neurological and neuro-otological disorders, suggesting an important unmet healthcare need for early diagnosis and targeted management strategies. Further studies are needed to determine the clinical associations and prognostic significance of radiologically defined iSS in the general population.
经典的幕下表面铁沉积症(iSS)被认为是一种罕见但可能导致残疾的神经系统疾病。它是由于慢性血液缓慢渗入脑脊液,以及铁降解产物含铁血黄素在中枢神经系统软膜下层沉积所致。磁敏感加权(SW)磁共振成像(MRI)是参考诊断方法。很少有研究描述小脑或幕下铁沉积症的人群患病率,英国人群中尚无此类研究。这项横断面观察性研究的目的是使用预先定义的放射学标准,估计英国生物银行样本中iSS的患病率。
我们回顾了英国生物银行参与者的SW MRI,寻找经典iSS的放射学特征:幕下结构(上蚓部、小脑叶片、脑干或颅颈交界区)受累。我们使用威尔逊评分公式,将点患病率计算为每审查病例总数中确诊病例数,并计算95%置信区间(CI)。
在审查的10305份SW MRI中,发现5例具有iSS放射学特征的病例,显示小脑/上蚓部受累。iSS的估计患病率计算为每10万人口中有48.5例(95%CI 20.7 - 113.5)。
这是第一项估计英国人群中iSS患病率的研究。iSS的患病率高于其他罕见神经系统和神经耳科疾病的报告患病率,这表明早期诊断和针对性管理策略存在重要的未满足医疗需求。需要进一步研究以确定一般人群中放射学定义的iSS的临床关联和预后意义。