Literature DB >> 26510000

BATTEN DISEASE CAUSED BY A NOVEL MUTATION IN THE PPT1 GENE.

Tatyana I Metelitsina1, Darrel J Waggoner, Michael A Grassi.   

Abstract

PURPOSE: To report a case of Batten disease due to a previously unreported mutation in PPT1.
METHODS: A 9-year-old girl presented with classic clinical findings of Batten Disease.
RESULTS: Genetic testing for the mutations in the most common Batten disease gene, CLN3, was negative. Evaluation of a panel of genes known to be implicated in neuronal ceroid lipofuscinoses revealed disease causing mutations in PPT1, one of which was novel.
CONCLUSION: Mutations in PPT1 typically cause the infantile form of neuronal ceroid lipofuscinosis. Clinical diagnosis of the juvenile form of neuronal ceroid lipofuscinosis, Batten disease, should still be considered in cases with negative CLN3 genetic testing. Batten disease can occur due to genetic heterogeneity. Testing of other members of the neuronal ceroid lipofuscinosis gene family can lead to confirmation of the correct diagnosis.

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Year:  2016        PMID: 26510000      PMCID: PMC4851602          DOI: 10.1097/ICB.0000000000000227

Source DB:  PubMed          Journal:  Retin Cases Brief Rep        ISSN: 1935-1089


  9 in total

1.  FINE STRUCTURE OF THE LIPID BODIES IN JUVENILE AMAUROTIC IDIOCY.

Authors:  W ZEMAN; S DONAHUE
Journal:  Acta Neuropathol       Date:  1963-11-05       Impact factor: 17.088

2.  Batten disease: features to facilitate early diagnosis.

Authors:  J Collins; G E Holder; H Herbert; G G W Adams
Journal:  Br J Ophthalmol       Date:  2006-06-05       Impact factor: 4.638

Review 3.  Correlations between genotype, ultrastructural morphology and clinical phenotype in the neuronal ceroid lipofuscinoses.

Authors:  Sara E Mole; Ruth E Williams; Hans H Goebel
Journal:  Neurogenetics       Date:  2005-09-28       Impact factor: 2.660

Review 4.  Classification and natural history of the neuronal ceroid lipofuscinoses.

Authors:  Jonathan W Mink; Erika F Augustine; Heather R Adams; Frederick J Marshall; Jennifer M Kwon
Journal:  J Child Neurol       Date:  2013-07-09       Impact factor: 1.987

Review 5.  The neuronal ceroid-lipofuscinoses: from past to present.

Authors:  Matti Haltia
Journal:  Biochim Biophys Acta       Date:  2006-07-08

6.  Mutations in the palmitoyl-protein thioesterase gene (PPT; CLN1) causing juvenile neuronal ceroid lipofuscinosis with granular osmiophilic deposits.

Authors:  H M Mitchison; S L Hofmann; C H Becerra; P B Munroe; B D Lake; Y J Crow; J B Stephenson; R E Williams; I L Hofman; P E Taschner; J J Martin; M Philippart; E Andermann; F Andermann; S E Mole; R M Gardiner; A M O'Rawe
Journal:  Hum Mol Genet       Date:  1998-02       Impact factor: 6.150

7.  Funduscopic and angiographic appearance in the neuronal ceroid lipofuscinoses.

Authors:  Dean P Hainsworth; Grace T Liu; Charles W Hamm; Martin L Katz
Journal:  Retina       Date:  2009-05       Impact factor: 4.256

Review 8.  The dystrophic retina in multisystem disorders: the electroretinogram in neuronal ceroid lipofuscinoses.

Authors:  R G Weleber
Journal:  Eye (Lond)       Date:  1998       Impact factor: 3.775

9.  Mutations in CLN7/MFSD8 are a common cause of variant late-infantile neuronal ceroid lipofuscinosis.

Authors:  Maria Kousi; Eija Siintola; Lenka Dvorakova; Hana Vlaskova; Julie Turnbull; Meral Topcu; Deniz Yuksel; Sarenur Gokben; Berge A Minassian; Milan Elleder; Sara E Mole; Anna-Elina Lehesjoki
Journal:  Brain       Date:  2009-02-05       Impact factor: 15.255

  9 in total
  3 in total

Review 1.  Autosomal dominant neuronal ceroid lipofuscinosis: Clinical features and molecular basis.

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Journal:  Clin Genet       Date:  2020-08-26       Impact factor: 4.438

2.  GNS561, a clinical-stage PPT1 inhibitor, is efficient against hepatocellular carcinoma via modulation of lysosomal functions.

Authors:  Sonia Brun; Eloïne Bestion; Eric Raymond; Firas Bassissi; Zuzana Macek Jilkova; Soraya Mezouar; Madani Rachid; Marie Novello; Jennifer Tracz; Ahmed Hamaï; Gilles Lalmanach; Lise Vanderlynden; Raphael Legouffe; Jonathan Stauber; Thomas Schubert; Maximilian G Plach; Jérôme Courcambeck; Cyrille Drouot; Guillaume Jacquemot; Cindy Serdjebi; Gael Roth; Jean-Pierre Baudoin; Christelle Ansaldi; Thomas Decaens; Philippe Halfon
Journal:  Autophagy       Date:  2021-11-05       Impact factor: 13.391

3.  Mice deficient in the lysosomal enzyme palmitoyl-protein thioesterase 1 (PPT1) display a complex retinal phenotype.

Authors:  Yevgeniya Atiskova; Susanne Bartsch; Tatyana Danyukova; Elke Becker; Christian Hagel; Stephan Storch; Udo Bartsch
Journal:  Sci Rep       Date:  2019-10-02       Impact factor: 4.379

  3 in total

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