Literature DB >> 19298904

Pseudoxanthoma elasticum: genetics, clinical manifestations and therapeutic approaches.

Robert P Finger1, Peter Charbel Issa, Markus S Ladewig, Christian Götting, Christina Szliska, Hendrik P N Scholl, Frank G Holz.   

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.

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Year:  2009        PMID: 19298904     DOI: 10.1016/j.survophthal.2008.12.006

Source DB:  PubMed          Journal:  Surv Ophthalmol        ISSN: 0039-6257            Impact factor:   6.048


  43 in total

Review 1.  [Clinical applications of OCT angiography].

Authors:  P P Fang; M Lindner; J S Steinberg; P L Müller; M Gliem; P Charbel Issa; T U Krohne; F G Holz
Journal:  Ophthalmologe       Date:  2016-01       Impact factor: 1.059

Review 2.  ABCC6 as a target in pseudoxanthoma elasticum.

Authors:  András Váradi; Zalán Szabó; Viola Pomozi; Hugues de Boussac; Krisztina Fülöp; Tamás Arányi
Journal:  Curr Drug Targets       Date:  2011-05       Impact factor: 3.465

3.  Oral warning signs of elastic pseudoxanthoma.

Authors:  Lara Passos Alvim; Thaís Izidoro Pires; Maria das Graças Afonso Miranda Chaves; Cynthia Savioli; Gisele Maria Campos Fabri
Journal:  Womens Health (Lond)       Date:  2016-09-15

4.  [Hereditary metabolic diseases with cutaneous manifestations : An update].

Authors:  J Frank; P Poblete-Gutiérrez
Journal:  Hautarzt       Date:  2011-02       Impact factor: 0.751

5.  Polypoidal choroidal vasculopathy in a patient with angioid streaks secondary to pseudoxanthoma elasticum.

Authors:  Stéphanie Baillif-Gostoli; Maddalena Quaranta-El Maftouhi; Martine Mauget-Faÿsse
Journal:  Graefes Arch Clin Exp Ophthalmol       Date:  2010-03-06       Impact factor: 3.117

6.  Pseudoxanthoma Elasticum: A Novel Mutation in the ABCC6 Gene That Affects Eye Manifestations of the Disease.

Authors:  Richard J Hesse; Joshua Groetsch; Alan Burshell
Journal:  Ochsner J       Date:  2010

7.  New insights into the roles of the N-terminal region of the ABCC6 transporter.

Authors:  Rocchina Miglionico; Andrea Gerbino; Angela Ostuni; Maria Francesca Armentano; Magnus Monné; Monica Carmosino; Faustino Bisaccia
Journal:  J Bioenerg Biomembr       Date:  2016-03-04       Impact factor: 2.945

8.  Intravitreal aflibercept for refractory choroidal neovascularization secondary to angioid streaks.

Authors:  M Tetikoglu; H M Sagdik; S Aktas; F Ozcura
Journal:  Eye (Lond)       Date:  2016-03-04       Impact factor: 3.775

9.  Dietary magnesium, not calcium, prevents vascular calcification in a mouse model for pseudoxanthoma elasticum.

Authors:  Theo G M F Gorgels; Jan H Waarsing; Anneke de Wolf; Jacoline B ten Brink; Willem J P Loves; Arthur A B Bergen
Journal:  J Mol Med (Berl)       Date:  2010-02-23       Impact factor: 4.599

Review 10.  Retinal abnormalities in β-thalassemia major.

Authors:  Devang L Bhoiwala; Joshua L Dunaief
Journal:  Surv Ophthalmol       Date:  2015-08-29       Impact factor: 6.048

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