Literature DB >> 20583119

Digital necroses and vascular thrombosis in severe spinal muscular atrophy.

Sabine Rudnik-Schöneborn1, Silke Vogelgesang, Sven Armbrust, Luitgard Graul-Neumann, Christoph Fusch, Klaus Zerres.   

Abstract

Infantile spinal muscular atrophy (SMA) caused by homozygous SMN1 gene deletions/mutations is characterized by neuronal loss and axonopathy of motor neurons. We report two unrelated patients with severe SMA type I who had only one SMN2 copy and developed ulcerations and necroses of the fingers and toes. Sural nerve biopsy was normal in patient 1, whose affected skin displayed necroses and thrombotic occlusions of small vessels. Corresponding to a mouse model and other patients with similar findings, we believe that severe survival motor neuron (SMN) deficiency may present as vasculopathy.

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Year:  2010        PMID: 20583119     DOI: 10.1002/mus.21654

Source DB:  PubMed          Journal:  Muscle Nerve        ISSN: 0148-639X            Impact factor:   3.217


  40 in total

1.  Partial restoration of cardio-vascular defects in a rescued severe model of spinal muscular atrophy.

Authors:  Monir Shababi; Javad Habibi; Lixin Ma; Jacqueline J Glascock; James R Sowers; Christian L Lorson
Journal:  J Mol Cell Cardiol       Date:  2012-01-17       Impact factor: 5.000

Review 2.  Spinal muscular atrophy: diagnosis and management in a new therapeutic era.

Authors:  W David Arnold; Darine Kassar; John T Kissel
Journal:  Muscle Nerve       Date:  2014-12-16       Impact factor: 3.217

3.  Systemic, postsymptomatic antisense oligonucleotide rescues motor unit maturation delay in a new mouse model for type II/III spinal muscular atrophy.

Authors:  Laurent P Bogdanik; Melissa A Osborne; Crystal Davis; Whitney P Martin; Andrew Austin; Frank Rigo; C Frank Bennett; Cathleen M Lutz
Journal:  Proc Natl Acad Sci U S A       Date:  2015-10-12       Impact factor: 11.205

4.  Genetic inhibition of JNK3 ameliorates spinal muscular atrophy.

Authors:  Naresh K Genabai; Saif Ahmad; Zhanying Zhang; Xiaoting Jiang; Cynthia A Gabaldon; Laxman Gangwani
Journal:  Hum Mol Genet       Date:  2015-09-30       Impact factor: 6.150

5.  Mutations in the survival motor neuron (SMN) protein alter the dynamic nature of nuclear bodies.

Authors:  Robert Morse; Adrian G Todd; Debra J Shaw; Alison L McConville; Iain M Robinson; Philip J Young
Journal:  Neuromolecular Med       Date:  2010-11-17       Impact factor: 3.843

6.  Increased IGF-1 in muscle modulates the phenotype of severe SMA mice.

Authors:  Marta Bosch-Marcé; Claribel D Wee; Tara L Martinez; Celeste E Lipkes; Dong W Choe; Lingling Kong; James P Van Meerbeke; Antonio Musarò; Charlotte J Sumner
Journal:  Hum Mol Genet       Date:  2011-02-16       Impact factor: 6.150

7.  Prolactin increases SMN expression and survival in a mouse model of severe spinal muscular atrophy via the STAT5 pathway.

Authors:  Faraz Farooq; Francisco Abadía Molina; Jeremiah Hadwen; Duncan MacKenzie; Luke Witherspoon; Matthew Osmond; Martin Holcik; Alex MacKenzie
Journal:  J Clin Invest       Date:  2011-07-25       Impact factor: 14.808

8.  Systemic peptide-mediated oligonucleotide therapy improves long-term survival in spinal muscular atrophy.

Authors:  Suzan M Hammond; Gareth Hazell; Fazel Shabanpoor; Amer F Saleh; Melissa Bowerman; James N Sleigh; Katharina E Meijboom; Haiyan Zhou; Francesco Muntoni; Kevin Talbot; Michael J Gait; Matthew J A Wood
Journal:  Proc Natl Acad Sci U S A       Date:  2016-09-12       Impact factor: 11.205

9.  The neuromuscular impact of symptomatic SMN restoration in a mouse model of spinal muscular atrophy.

Authors:  W Arnold; Vicki L McGovern; Benjamin Sanchez; Jia Li; Kaitlyn M Corlett; Stephen J Kolb; Seward B Rutkove; Arthur H Burghes
Journal:  Neurobiol Dis       Date:  2015-12-28       Impact factor: 5.996

Review 10.  Spinal muscular atrophy: a motor neuron disorder or a multi-organ disease?

Authors:  Monir Shababi; Christian L Lorson; Sabine S Rudnik-Schöneborn
Journal:  J Anat       Date:  2013-07-22       Impact factor: 2.610

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