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弹性假黄瘤:遗传学、临床表现及治疗方法

Pseudoxanthoma elasticum: genetics, clinical manifestations and therapeutic approaches.

作者信息

Finger Robert P, Charbel Issa Peter, Ladewig Markus S, Götting Christian, Szliska Christina, Scholl Hendrik P N, Holz Frank G

机构信息

Department of Ophthalmology, University of Bonn, Bonn, Germany.

出版信息

Surv Ophthalmol. 2009 Mar-Apr;54(2):272-85. doi: 10.1016/j.survophthal.2008.12.006.

Abstract

Pseudoxanthoma elasticum (PXE) is an inherited disorder that is associated with accumulation of mineralized and fragmented elastic fibers in the skin, vascular walls, and Bruch's membrane in the eye. Clinically, patients exhibit characteristic lesions of the posterior segment of the eye including peau d'orange, angioid streaks, and choroidal neovascularisations, of the skin including soft, ivory-colored papules in a reticular pattern that predominantly affect the neck and large flexor surfaces, and of the cardiovascular system with peripheral and coronary arterial occlusive disease as well as gastrointestinal bleedings. There is yet no definitive therapy. Recent studies suggest that PXE is inherited almost exclusively as an autosomal recessive trait. Its prevalence has been estimated to be 1:25,000-100,000. Very recently, the ABCC6 gene on chromosome 16p13.1 was found to be associated with the disease. Mutations within ABCC6 cause reduced or absent transmembraneous transport that leads to accumulation of extracellular material. Presumably, this mechanism causes calcification of elastic fibers. Despite the characteristic clinical features, the variability in phenotypic expressions, and the low prevalence may be responsible for the disease being underdiagnosed. This review compiles and summarizes current knowledge of PXE pathogenesis and clinical findings. Furthermore, different therapeutic strategies to treat retinal manifestations are discussed, including thermal laser coagulation, photodynamic therapy, and intravitreal injections of drugs inhibiting vascular endothelial growth factor.

摘要

弹性假黄瘤(PXE)是一种遗传性疾病,与皮肤、血管壁和眼部布鲁赫膜中矿化和碎片化弹性纤维的积累有关。临床上,患者表现出眼部后段的特征性病变,包括橘皮样改变、血管样条纹和脉络膜新生血管;皮肤病变包括呈网状分布的柔软、象牙色丘疹,主要影响颈部和大的屈肌表面;心血管系统病变包括外周和冠状动脉闭塞性疾病以及胃肠道出血。目前尚无确切的治疗方法。最近的研究表明,PXE几乎完全以常染色体隐性性状遗传。其患病率估计为1:25,000 - 100,000。最近,发现位于16号染色体p13.1上的ABCC6基因与该疾病有关。ABCC6基因内的突变导致跨膜转运减少或缺失,从而导致细胞外物质积累。据推测,这种机制导致弹性纤维钙化。尽管有特征性的临床特征,但表型表达的变异性和低患病率可能导致该疾病诊断不足。本综述汇编并总结了目前关于PXE发病机制和临床发现的知识。此外,还讨论了治疗视网膜表现的不同治疗策略,包括热激光凝固、光动力疗法和玻璃体内注射抑制血管内皮生长因子的药物。

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