De Waele Liesbeth, Lagae Lieven, Mekahli Djalila
Department of Pediatric Neurology, University Hospitals Leuven, Herestraat 49, 3000, Leuven, Belgium,
Pediatr Nephrol. 2015 Oct;30(10):1771-80. doi: 10.1007/s00467-014-3027-9. Epub 2014 Dec 23.
Renal lesions represent the second most significant cause of morbidity and mortality in patients with tuberous sclerosis complex (TSC). Recent advances in the understanding of the pathophysiology of TSC have led to the exploration of new potential therapeutic targets. Clinical trials with mammalian target of rapamycin (mTOR) inhibitors have demonstrated promising results for several indications, such as renal angiomyolipoma, subependymal giant cell astrocytoma, lymphangioleiomyomatosis and facial angiofibromas. Currently, there is a scarcity of natural history data and randomized, placebo-controlled clinical trials on TSC. Recently, however, recommendations for the diagnostic criteria, surveillance, and management of TSC patients have been updated. This review focuses on these novel recommendations and highlights the need for multidisciplinary follow-up of this multi-systemic disease.
肾损害是结节性硬化症(TSC)患者发病和死亡的第二大重要原因。对TSC病理生理学认识的最新进展促使人们探索新的潜在治疗靶点。使用雷帕霉素哺乳动物靶点(mTOR)抑制剂的临床试验已在多个适应症上显示出有前景的结果,如肾血管平滑肌脂肪瘤、室管膜下巨细胞星形细胞瘤、淋巴管平滑肌瘤病和面部血管纤维瘤。目前,关于TSC的自然病史数据以及随机、安慰剂对照临床试验较为匮乏。然而,最近,TSC患者的诊断标准、监测和管理建议已得到更新。本综述重点关注这些新建议,并强调对这种多系统疾病进行多学科随访的必要性。