Literature DB >> 15851732

Cystic leukoencephalopathy without megalencephaly: a distinct disease entity in 15 children.

M Henneke1, N Preuss, V Engelbrecht, F Aksu, E Bertini, G Bibat, K Brockmann, C Hübner, M Mayer, V Mejaski-Bosnjak, S Naidu, E Neumaier-Probst, D Rodriguez, W Weisz, A Kohlschütter, J Gärtner.   

Abstract

OBJECTIVE: To describe a distinctive syndrome of nonprogressive encephalopathy, normo- or microcephaly, and early onset of severe psychomotor impairment in 15 white patients, including two siblings and two first cousins. METHODS AND
RESULTS: MRI revealed bilateral cysts in the anterior part of the temporal lobe and white matter abnormalities with pericystic abnormal myelination and symmetric lesions in frontal and occipital periventricular regions. None of the usual inborn errors of metabolism/infectious diseases associated with leukoencephalopathy and bilateral anterior temporal lobe cysts were detected.
CONCLUSIONS: These patients' clinical signs and cranial MRI abnormalities are strikingly similar and may represent a distinctive disease with autosomal-recessive inheritance: cystic leukoencephalopathy without megalencephaly.

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Year:  2005        PMID: 15851732     DOI: 10.1212/01.WNL.0000158472.82823.01

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  6 in total

Review 1.  Magnetic resonance imaging of anterior temporal lobe cysts in children: discriminating special imaging features in a particular group of diseases.

Authors:  Renato Hoffmann Nunes; Felipe Torres Pacheco; Antonio Jose da Rocha
Journal:  Neuroradiology       Date:  2014-04-23       Impact factor: 2.804

2.  RNASET2-deficient cystic leukoencephalopathy resembles congenital cytomegalovirus brain infection.

Authors:  Marco Henneke; Simone Diekmann; Andreas Ohlenbusch; Jens Kaiser; Volkher Engelbrecht; Alfried Kohlschütter; Ralph Krätzner; Marcos Madruga-Garrido; Michèle Mayer; Lennart Opitz; Diana Rodriguez; Franz Rüschendorf; Johannes Schumacher; Holger Thiele; Sven Thoms; Robert Steinfeld; Peter Nürnberg; Jutta Gärtner
Journal:  Nat Genet       Date:  2009-06-14       Impact factor: 38.330

3.  RNASET2-deficient leukoencephalopathy mimicking congenital CMV infection and Aicardi-Goutieres syndrome: a case report with a novel pathogenic variant.

Authors:  Reyhaneh Kameli; Man Amanat; Zahra Rezaei; Sareh Hosseionpour; Sedigheh Nikbakht; Houman Alizadeh; Mahmoud Reza Ashrafi; Abdolmajid Omrani; Masoud Garshasbi; Ali Reza Tavasoli
Journal:  Orphanet J Rare Dis       Date:  2019-07-26       Impact factor: 4.123

4.  Interferon-driven brain phenotype in a mouse model of RNaseT2 deficient leukoencephalopathy.

Authors:  Matthias Kettwig; Katharina Ternka; Kristin Wendland; Dennis Manfred Krüger; Silvia Zampar; Charlotte Schob; Jonas Franz; Abhishek Aich; Anne Winkler; M Sadman Sakib; Lalit Kaurani; Robert Epple; Hauke B Werner; Samy Hakroush; Julia Kitz; Marco Prinz; Eva Bartok; Gunther Hartmann; Simone Schröder; Peter Rehling; Marco Henneke; Susann Boretius; A Alia; Oliver Wirths; Andre Fischer; Christine Stadelmann; Stefan Nessler; Jutta Gärtner
Journal:  Nat Commun       Date:  2021-11-11       Impact factor: 14.919

5.  Zebrafish disease model of human RNASET2-deficient cystic leukoencephalopathy displays abnormalities in early microglia.

Authors:  Thomas Weber; Lars Schlotawa; Roland Dosch; Noémie Hamilton; Jens Kaiser; Stina Schiller; Britta Wenske; Jutta Gärtner; Marco Henneke
Journal:  Biol Open       Date:  2020-05-07       Impact factor: 2.422

6.  RNaseT2 knockout rats exhibit hippocampal neuropathology and deficits in memory.

Authors:  Kerstin W Sinkevicius; Thomas R Morrison; Praveen Kulkarni; Martha K Caffrey Cagliostro; Sade Iriah; Samantha Malmberg; Julia Sabrick; Jennifer A Honeycutt; Kim L Askew; Malav Trivedi; Craig F Ferris
Journal:  Dis Model Mech       Date:  2018-06-27       Impact factor: 5.758

  6 in total

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