Department of Neurology, Northwestern University, Ann and Robert H. Lurie Children's Hospital of Chicago, Chicago, Illinois.
Department of Pediatrics and Neurology, Wayne State University, Children's Hospital of Michigan, Detroit, Michigan.
Pediatr Neurol. 2018 Jul;84:11-20. doi: 10.1016/j.pediatrneurol.2018.04.005. Epub 2018 Apr 18.
Sturge-Weber syndrome is a neurocutaneous disorder associated with port-wine birthmark, leptomeningeal capillary malformations, and glaucoma. It is associated with an unpredictable clinical course. Because of its rarity and complexity, many physicians are unaware of the disease and its complications. A major focus moving ahead will be to turn knowledge gaps and unmet needs into new research directions.
On October 1-3, 2017, the Sturge-Weber Foundation assembled clinicians from the Clinical Care Network with patients from the Patient Engagement Network of the Sturge-Weber Foundation to identify our current state of knowledge, knowledge gaps, and unmet needs.
One clear unmet need is a need for consensus guidelines on care and surveillance. It was strongly recommended that patients be followed by multidisciplinary clinical teams with life-long follow-up for children and adults to monitor disease progression in the skin, eye, and brain. Standardized neuroimaging modalities at specified time points are needed together with a stronger clinicopathologic understanding. Uniform tissue banking and clinical data acquisition strategies are needed with cross-center, longitudinal studies that will set the stage for new clinical trials. A better understanding of the pathogenic roles of cerebral calcifications and stroke-like symptoms is a clear unmet need with potentially devastating consequences.
Biomarkers capable of predicting disease progression will be needed to advance new therapeutic strategies. Importantly, how to deal with the emotional and psychological effects of Sturge-Weber syndrome and its impact on quality of life is a clear unmet need.
Sturge-Weber 综合征是一种神经皮肤疾病,与葡萄酒色斑、软脑膜毛细血管畸形和青光眼有关。它具有不可预测的临床病程。由于其罕见性和复杂性,许多医生并不了解这种疾病及其并发症。未来的一个主要重点将是把知识空白和未满足的需求转化为新的研究方向。
2017 年 10 月 1 日至 3 日,Sturge-Weber 基金会召集了来自临床护理网络的临床医生和来自 Sturge-Weber 基金会患者参与网络的患者,以确定我们目前的知识状况、知识空白和未满足的需求。
一个明显的未满足需求是需要制定关于护理和监测的共识指南。强烈建议患者由多学科临床团队进行随访,对儿童和成人进行终身随访,以监测皮肤、眼睛和大脑的疾病进展。需要在特定时间点使用标准化的神经影像学模式,并结合更强的临床病理理解。需要统一的组织库和临床数据采集策略,进行跨中心、纵向研究,为新的临床试验奠定基础。更好地了解脑钙化和类似中风症状的致病作用是一个明显的未满足需求,可能会带来毁灭性的后果。
需要能够预测疾病进展的生物标志物来推进新的治疗策略。重要的是,如何应对 Sturge-Weber 综合征的情绪和心理影响及其对生活质量的影响是一个明显的未满足需求。