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Statements

Subject Item
n2:RIV%2F61989592%3A15120%2F14%3A33149356%21RIV15-MSM-15120___
rdf:type
n17:Vysledek skos:Concept
dcterms:description
In patients with Wilson's disease (WD) transcranial brain sonography typically reveals areas of increased echogenicity (hyperechogenicity) of the lenticular nucleus (LN). Correlation with T2-hypointensity on magnetic resonance images suggested that LN hyperechogenicity in WD is caused by trace metal accumulation. Accumulation of both, copper and iron, in the brain of WD patients has been reported. The present study was designed to elucidate whether LN hyperechogenicity in WD reflects accumulation of copper or iron. Post-mortem brains of 15 WD patients and one non-WD subject were studied with ultrasonography in an investigator-blinded fashion. LN hyperechogenicity was measured planimetrically by manual tracing as well as using digitized image analysis. The putaminal copper content was determined in samples of 11 WD brains and the non-WD brains using inductively coupled plasma mass spectrometry, and iron content was assessed using flame atomic absorption spectroscopy. LN was normal on ultrasonography only in the non-WD brain, but abnormal (hyperechogenic) in all WD brains. Digitized image analysis measures of LN hyperechogenicity and, by trend, manual measures correlated with putaminal copper content (Pearson test; digitized: r = 0.77, p = 0.04; manual: r = 0.57, p = 0.051) but not with iron content (each, p } 0.18). LN hyperechogenicity measures were unrelated to age at death of patients, age at onset of WD, WD duration, age of brain specimen, serum copper or serum ceruloplasmin (each, p } 0.1). We conclude that LN hyperechogenicity in WD reflects copper, but not iron accumulation. Further studies are warranted to elucidate the use of transcranial brain sonography for monitoring therapeutic effects of chelating agents in WD patients. In patients with Wilson's disease (WD) transcranial brain sonography typically reveals areas of increased echogenicity (hyperechogenicity) of the lenticular nucleus (LN). Correlation with T2-hypointensity on magnetic resonance images suggested that LN hyperechogenicity in WD is caused by trace metal accumulation. Accumulation of both, copper and iron, in the brain of WD patients has been reported. The present study was designed to elucidate whether LN hyperechogenicity in WD reflects accumulation of copper or iron. Post-mortem brains of 15 WD patients and one non-WD subject were studied with ultrasonography in an investigator-blinded fashion. LN hyperechogenicity was measured planimetrically by manual tracing as well as using digitized image analysis. The putaminal copper content was determined in samples of 11 WD brains and the non-WD brains using inductively coupled plasma mass spectrometry, and iron content was assessed using flame atomic absorption spectroscopy. LN was normal on ultrasonography only in the non-WD brain, but abnormal (hyperechogenic) in all WD brains. Digitized image analysis measures of LN hyperechogenicity and, by trend, manual measures correlated with putaminal copper content (Pearson test; digitized: r = 0.77, p = 0.04; manual: r = 0.57, p = 0.051) but not with iron content (each, p } 0.18). LN hyperechogenicity measures were unrelated to age at death of patients, age at onset of WD, WD duration, age of brain specimen, serum copper or serum ceruloplasmin (each, p } 0.1). We conclude that LN hyperechogenicity in WD reflects copper, but not iron accumulation. Further studies are warranted to elucidate the use of transcranial brain sonography for monitoring therapeutic effects of chelating agents in WD patients.
dcterms:title
Lenticular nucleus hyperechogenicity in Wilson's disease reflects local copper, but not iron accumulation Lenticular nucleus hyperechogenicity in Wilson's disease reflects local copper, but not iron accumulation
skos:prefLabel
Lenticular nucleus hyperechogenicity in Wilson's disease reflects local copper, but not iron accumulation Lenticular nucleus hyperechogenicity in Wilson's disease reflects local copper, but not iron accumulation
skos:notation
RIV/61989592:15120/14:33149356!RIV15-MSM-15120___
n3:aktivita
n9:S
n3:aktivity
S
n3:cisloPeriodika
121 (10)
n3:dodaniDat
n16:2015
n3:domaciTvurceVysledku
n4:3054691
n3:druhVysledku
n6:J
n3:duvernostUdaju
n18:S
n3:entitaPredkladatele
n7:predkladatel
n3:idSjednocenehoVysledku
25979
n3:idVysledku
RIV/61989592:15120/14:33149356
n3:jazykVysledku
n14:eng
n3:klicovaSlova
Wilson's disease; iron; copper; transcranial sonography; lenticular nucleus
n3:klicoveSlovo
n10:transcranial%20sonography n10:Wilson%27s%20disease n10:lenticular%20nucleus n10:iron n10:copper
n3:kodStatuVydavatele
AT - Rakouská republika
n3:kontrolniKodProRIV
[2390CB9F9216]
n3:nazevZdroje
Journal of Neural Transmission
n3:obor
n12:FP
n3:pocetDomacichTvurcuVysledku
1
n3:pocetTvurcuVysledku
8
n3:rokUplatneniVysledku
n16:2014
n3:svazekPeriodika
2014
n3:tvurceVysledku
Czlonkowska, Anna Szpak, Grazyna Maria Bulska, Ewa Litwin, Tomasz Walter, Uwe Jablonka-Salach, Katarzina Školoudík, David Skowrońska, Marta
s:issn
0300-9564
s:numberOfPages
7
n15:doi
10.1007/s00702-014-1184-4
n13:organizacniJednotka
15120