Literature DB >> 27445018

Unique Characteristics of the Photoparoxysmal Response in Patients With Neuronal Ceroid Lipofuscinosis Type 2: Can EEG Be a Biomarker?

Dara V Albert1, Han Yin2, Emily C De Los Reyes3, Jorge Vidaurre3.   

Abstract

OBJECTIVE: The objective was to identify unique features of the photoparoxysmal response seen in patients with neuronal ceroid lipofuscinosis type 2 as compared to patients with a photoparoxysmal response associated with other epilepsy syndromes.
METHODS: Electroencephalograms from patients with neuronal ceroid lipofuscinosis type 2 seen at the authors' institution in the past 10 years as well as electroencephalograms (EEGs) reported to have a photoparoxysmal response during a single year were reviewed.
RESULTS: A photoparoxysmal response was seen in 60% of the patients with neuronal ceroid lipofuscinosis type 2. This was most commonly seen with low frequency intermittent photic stimulation (76%) which often occurred in a time-locked fashion (63%) and was seen on the patient's initial EEG (78%). A unique pattern the authors called "sentinel" discharge was identified in 30% of EEGs in patients with neuronal ceroid lipofuscinosis.
CONCLUSIONS: Photoparoxysmal responses in patients with neuronal ceroid lipofuscinosis type 2 have features which are distinguishing from photoparoxysmal responses seen in other epilepsies.
© The Author(s) 2016.

Entities:  

Keywords:  electroencephalogram (EEG); neuronal ceroid lipofuscinosis; pediatric epilepsy; photic stimulation; photoparoxysmal response

Mesh:

Year:  2016        PMID: 27445018     DOI: 10.1177/0883073816658659

Source DB:  PubMed          Journal:  J Child Neurol        ISSN: 0883-0738            Impact factor:   1.987


  7 in total

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Authors:  Konrad Kaminiów; Sylwia Kozak; Justyna Paprocka
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2.  Neuronal ceroid lipofuscinosis type 2: an Australian case series.

Authors:  Alexandra M Johnson; Simone Mandelstam; Ian Andrews; Katja Boysen; Joy Yaplito-Lee; Michael Fietz; Lakshmi Nagarajan; Victoria Rodriguez-Casero; Monique M Ryan; Nicholas Smith; Ingrid E Scheffer; Carolyn Ellaway
Journal:  J Paediatr Child Health       Date:  2020-04-24       Impact factor: 1.954

3.  Guidelines on the diagnosis, clinical assessments, treatment and management for CLN2 disease patients.

Authors:  Sara E Mole; Angela Schulz; Eben Badoe; Samuel F Berkovic; Emily C de Los Reyes; Simon Dulz; Paul Gissen; Norberto Guelbert; Charles M Lourenco; Heather L Mason; Jonathan W Mink; Noreen Murphy; Miriam Nickel; Joffre E Olaya; Maurizio Scarpa; Ingrid E Scheffer; Alessandro Simonati; Nicola Specchio; Ina Von Löbbecke; Raymond Y Wang; Ruth E Williams
Journal:  Orphanet J Rare Dis       Date:  2021-04-21       Impact factor: 4.123

Review 4.  Neurophysiological Findings in Neuronal Ceroid Lipofuscinoses.

Authors:  Marina Trivisano; Alessandro Ferretti; Costanza Calabrese; Nicola Pietrafusa; Ludovica Piscitello; Giusy Carfi' Pavia; Federico Vigevano; Nicola Specchio
Journal:  Front Neurol       Date:  2022-02-25       Impact factor: 4.003

5.  Genetic and phenotypic spectrum of Chinese patients with epilepsy and photosensitivity.

Authors:  Yue Niu; Pan Gong; Xianru Jiao; Zhao Xu; Yuehua Zhang; Zhixian Yang
Journal:  Front Neurol       Date:  2022-08-02       Impact factor: 4.086

6.  Targeted re-sequencing for early diagnosis of genetic causes of childhood epilepsy: the Italian experience from the 'beyond epilepsy' project.

Authors:  Elisabetta Amadori; Marcello Scala; Giulia Sofia Cereda; Maria Stella Vari; Francesca Marchese; Veronica Di Pisa; Maria Margherita Mancardi; Thea Giacomini; Laura Siri; Fabiana Vercellino; Domenico Serino; Alessandro Orsini; Alice Bonuccelli; Irene Bagnasco; Amanda Papa; Carlo Minetti; Duccio Maria Cordelli; Pasquale Striano
Journal:  Ital J Pediatr       Date:  2020-07-06       Impact factor: 3.288

7.  An ERG and OCT study of neuronal ceroid lipofuscinosis CLN2 Battens retinopathy.

Authors:  Dorothy A Thompson; Siân E Handley; Robert H Henderson; Oliver R Marmoy; Paul Gissen
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  7 in total

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