Megalencephalic leukoencephalopathy with subcortical cysts: chronic white matter oedema due to a defect in brain ion and water homoeostasis

MS van der Knaap, I Boor, R Estévez - The Lancet Neurology, 2012 - thelancet.com
Megalencephalic leukoencephalopathy with subcortical cysts (MLC) is characterised by
chronic white matter oedema. The disease has an infantile onset and leads to slow …

Exon-skipping antisense oligonucleotides to correct missplicing in neurogenetic diseases

K Siva, G Covello, MA Denti - Nucleic acid therapeutics, 2014 - liebertpub.com
Alternative splicing is an important regulator of the transcriptome. However, mutations may
cause alteration of splicing patterns, which in turn leads to disease. During the past 10 …

Structure-guided combination therapy to potently improve the function of mutant CFTRs

G Veit, H Xu, E Dreano, RG Avramescu, M Bagdany… - Nature medicine, 2018 - nature.com
Available corrector drugs are unable to effectively rescue the folding defects of CFTR-ΔF508
(or CFTR-F508del), the most common disease-causing mutation of the cystic fibrosis …

Investigation of LRRC8-mediated volume-regulated anion currents in Xenopus oocytes

H Gaitán-Peñas, A Gradogna, L Laparra-Cuervo… - Biophysical journal, 2016 - cell.com
Volume-regulated anion channels (VRACs) play an important role in controlling cell volume
by opening upon cell swelling. Recent work has shown that heteromers of LRRC8A with …

Mutant GlialCAM causes megalencephalic leukoencephalopathy with subcortical cysts, benign familial macrocephaly, and macrocephaly with retardation and autism

T López-Hernández, MC Ridder, M Montolio… - The American Journal of …, 2011 - cell.com
Megalencephalic leukoencephalopathy with subcortical cysts (MLC) is a leukodystrophy
characterized by early-onset macrocephaly and delayed-onset neurological deterioration …

GlialCAM, a protein defective in a leukodystrophy, serves as a ClC-2 Cl− channel auxiliary subunit

E Jeworutzki, T López-Hernández, X Capdevila-Nortes… - Neuron, 2012 - cell.com
Ion fluxes mediated by glial cells are required for several physiological processes such as
fluid homeostasis or the maintenance of low extracellular potassium during high neuronal …

Molecular mechanisms of MLC1 and GLIALCAM mutations in megalencephalic leukoencephalopathy with subcortical cysts

T López-Hernández, S Sirisi… - Human molecular …, 2011 - academic.oup.com
Megalencephalic leukoencephalopathy with subcortical cysts (MLC) is a rare
leukodystrophy caused by mutations in MLC1 or GLIALCAM. The GLIALCAM gene product …

Leukodystrophies: classification, diagnosis, and treatment

DJ Costello, AF Eichler, FS Eichler - The neurologist, 2009 - journals.lww.com
Background: The leukodystrophies are a heterogeneous group of diseases, which primarily
affect white matter. Symptomatic patients are frequently misdiagnosed and the …

Mice with megalencephalic leukoencephalopathy with cysts: a developmental angle

M Dubey, M Bugiani, MC Ridder, NL Postma… - Annals of …, 2015 - Wiley Online Library
Objective Megalencephalic leukoencephalopathy with cysts (MLC) is a genetic disease
characterized by infantile onset white matter edema and delayed onset neurological …

Protein quality control at the plasma membrane

T Okiyoneda, PM Apaja, GL Lukacs - Current opinion in cell biology, 2011 - Elsevier
Cellular proteostasis (or protein homeostasis) depends on the timely folding and disposal of
conformationally damaged polypeptides during their life span at all subcellular locations …