Literature DB >> 15715047

A variant form of neuronal ceroid lipofuscinosis in American bulldogs.

Jason Evans1, Martin L Katz, Donald Levesque, G Diane Shelton, Alexander de Lahunta, Dennis O'Brien.   

Abstract

Neuronal ceroid lipofuscinoses (NCLs) are inherited neurodegenerative diseases characterized by accumulations of autofluorescent lipopigments within cells of the nervous system. Nine related American Bulldogs demonstrated dysmetria in all limbs and paraparesis. Nonambulatory tetraparesis was observed only in the later stages of the disease. The clinical signs developed between 1 and 3 years of age and were slowly progressive over several years, which is inconsistent with most reports in other breeds. Results from blood tests for 8 different lysosomal storage diseases on 4 affected and 6 related but unaffected dogs were negative. Four affected dogs were euthanized and histopathologic examinations showed diffuse accumulations of periodic acid-Schiff-positive inclusions in neurons and axonal spheroids along the entire neuraxis and retinae. The most severe lesions were in the brainstem proprioceptive nuclei and spinal cord, consistent with clinical signs. The storage material was autofluorescent and immunohistochemically positive for products of lipid peroxidation. Ultrastructural analysis was consistent with NCL. Pedigree analysis supports an autosomal-recessive mode of inheritance. NCL has not been previously reported in the American Bulldog and these findings suggest a variant form of the canine disease.

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Year:  2005        PMID: 15715047     DOI: 10.1892/0891-6640(2005)19<44:avfonc>2.0.co;2

Source DB:  PubMed          Journal:  J Vet Intern Med        ISSN: 0891-6640            Impact factor:   3.333


  7 in total

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Authors:  Krystal Weber; David A Pearce
Journal:  J Child Neurol       Date:  2013-09       Impact factor: 1.987

2.  A mixed breed dog with neuronal ceroid lipofuscinosis is homozygous for a CLN5 nonsense mutation previously identified in Border Collies and Australian Cattle Dogs.

Authors:  Natalie A Villani; Garrett Bullock; Jennifer R Michaels; Osamu Yamato; Dennis P O'Brien; Tendai Mhlanga-Mutangadura; Gary S Johnson; Martin L Katz
Journal:  Mol Genet Metab       Date:  2019-04-17       Impact factor: 4.797

Review 3.  Canine epilepsy genetics.

Authors:  Kari J Ekenstedt; Edward E Patterson; James R Mickelson
Journal:  Mamm Genome       Date:  2011-10-30       Impact factor: 2.957

4.  A mutation in canine PPT1 causes early onset neuronal ceroid lipofuscinosis in a Dachshund.

Authors:  Douglas N Sanders; Fabiana H Farias; Gary S Johnson; Vivian Chiang; James R Cook; Dennis P O'Brien; Sandra L Hofmann; Jui-Yun Lu; Martin L Katz
Journal:  Mol Genet Metab       Date:  2010-04-24       Impact factor: 4.797

5.  Screening and Carrier Rate of Neuronal Ceroid Lipofuscinosis in Chihuahua Dogs in Japan.

Authors:  Shahnaj Pervin; Md Shafiqul Islam; Naomi Tada; Toshihiko Tsutsui; Mohammad Mahbubur Rahman; Akira Yabuki; Martia Rani Tacharina; Tofazzal Md Rakib; Shinichiro Maki; Osamu Yamato
Journal:  Animals (Basel)       Date:  2022-05-07       Impact factor: 3.231

6.  Homozygous PPT1 Splice Donor Mutation in a Cane Corso Dog With Neuronal Ceroid Lipofuscinosis.

Authors:  A Kolicheski; H L Barnes Heller; S Arnold; R D Schnabel; J F Taylor; C A Knox; T Mhlanga-Mutangadura; D P O'Brien; G S Johnson; J Dreyfus; M L Katz
Journal:  J Vet Intern Med       Date:  2016-12-23       Impact factor: 3.333

Review 7.  Ferroptosis and Its Modulation by Autophagy in Light of the Pathogenesis of Lysosomal Storage Diseases.

Authors:  Karolina Pierzynowska; Estera Rintz; Lidia Gaffke; Grzegorz Węgrzyn
Journal:  Cells       Date:  2021-02-10       Impact factor: 6.600

  7 in total

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