Literature DB >> 20454899

[Diagnostics and treatment of neuronal ceroid lipofuscinoses from the viewpoint of neuropediatricians].

R Steinfeld1.   

Abstract

The neuronal ceroid lipofuscinoses (NCL) are a heterogeneous group of lysosomal diseases with rapidly progressive neurodegeneration and characteristic lipopigmentary lysosomal inclusions. The clinical picture is characterized by motor disturbances, developmental delay, behavioral abnormalities, epilepsy, loss of vision and dementia. Cranial MRI reveals global brain atrophy and in particular early atrophy of the cerebellum. If an NCL disease is suspected initial diagnostic assessment for the CLN1, CLN2, CLN3 and CLN10 subtypes is recommended. The investigations can be done with a dried blood spotted on filter paper. If the results are negative but an NCL disease is still suspected the further approach should be coordinated with an expert in the field. Possible other diagnostic examinations include electron microscopy of the storage material in lymphocytes and skin biopsy specimens or molecular genetic analysis of the suspected NCL gene. At present only symptomatic therapy is available for NCL diseases.

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Year:  2010        PMID: 20454899     DOI: 10.1007/s00347-009-2109-8

Source DB:  PubMed          Journal:  Ophthalmologe        ISSN: 0941-293X            Impact factor:   1.059


  9 in total

1.  Biochemical analysis of mutations in palmitoyl-protein thioesterase causing infantile and late-onset forms of neuronal ceroid lipofuscinosis.

Authors:  A K Das; J Y Lu; S L Hofmann
Journal:  Hum Mol Genet       Date:  2001-06-15       Impact factor: 6.150

2.  Spectrum of mutations in the Batten disease gene, CLN3.

Authors:  P B Munroe; H M Mitchison; A M O'Rawe; J W Anderson; R M Boustany; T J Lerner; P E Taschner; N de Vos; M H Breuning; R M Gardiner; S E Mole
Journal:  Am J Hum Genet       Date:  1997-08       Impact factor: 11.025

3.  Cathepsin D deficiency is associated with a human neurodegenerative disorder.

Authors:  Robert Steinfeld; Konstanze Reinhardt; Kathrin Schreiber; Merle Hillebrand; Ralph Kraetzner; Wolfgang Bruck; Paul Saftig; Jutta Gartner
Journal:  Am J Hum Genet       Date:  2006-03-29       Impact factor: 11.025

4.  The gene mutated in variant late-infantile neuronal ceroid lipofuscinosis (CLN6) and in nclf mutant mice encodes a novel predicted transmembrane protein.

Authors:  Ruth B Wheeler; Julie D Sharp; Roger A Schultz; John M Joslin; Ruth E Williams; Sara E Mole
Journal:  Am J Hum Genet       Date:  2001-11-27       Impact factor: 11.025

5.  Cathepsin D deficiency underlies congenital human neuronal ceroid-lipofuscinosis.

Authors:  Eija Siintola; Sanna Partanen; Petter Strömme; Aleksi Haapanen; Matti Haltia; Jan Maehlen; Anna-Elina Lehesjoki; Jaana Tyynelä
Journal:  Brain       Date:  2006-05-02       Impact factor: 13.501

6.  Mutations in classical late infantile neuronal ceroid lipofuscinosis disrupt transport of tripeptidyl-peptidase I to lysosomes.

Authors:  Robert Steinfeld; Hans-Bertram Steinke; Dirk Isbrandt; Alfried Kohlschütter; Jutta Gärtner
Journal:  Hum Mol Genet       Date:  2004-08-18       Impact factor: 6.150

7.  The novel neuronal ceroid lipofuscinosis gene MFSD8 encodes a putative lysosomal transporter.

Authors:  Eija Siintola; Meral Topcu; Nina Aula; Hannes Lohi; Berge A Minassian; Andrew D Paterson; Xiao-Qing Liu; Callum Wilson; Ulla Lahtinen; Anna-Kaisa Anttonen; Anna-Elina Lehesjoki
Journal:  Am J Hum Genet       Date:  2007-05-14       Impact factor: 11.025

8.  Variant late infantile neuronal ceroid lipofuscinosis in a subset of Turkish patients is allelic to Northern epilepsy.

Authors:  Susanna Ranta; Meral Topcu; Saara Tegelberg; Hüseyin Tan; Alp Ustübütün; Isil Saatci; Andreas Dufke; Herbert Enders; Keith Pohl; Yves Alembik; Wayne A Mitchell; Sara E Mole; Anna-Elina Lehesjoki
Journal:  Hum Mutat       Date:  2004-04       Impact factor: 4.878

9.  CLN5, a novel gene encoding a putative transmembrane protein mutated in Finnish variant late infantile neuronal ceroid lipofuscinosis.

Authors:  M Savukoski; T Klockars; V Holmberg; P Santavuori; E S Lander; L Peltonen
Journal:  Nat Genet       Date:  1998-07       Impact factor: 38.330

  9 in total
  2 in total

1.  Congenital CLN disease in two siblings.

Authors:  Sascha Meyer; Umut Yilmaz; Yoo-Jin Kim; Robert Steinfeld; Gabriele Meyberg-Solomayer; Barbara Oehl-Jaschkowitz; Andreas Tzschach; Ludwig Gortner; Julia Igel; Otto Schofer
Journal:  Wien Med Wochenschr       Date:  2015-06-10

2.  [Neurological alterations and intellectual deficits with sudden visual loss in a 7-year-old boy].

Authors:  A Gotz-Wieckowska; M Pawlak; J Siwiec-Proscinska; M Seget
Journal:  Ophthalmologe       Date:  2013-05       Impact factor: 1.059

  2 in total

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