Literature DB >> 35927425

Spinal muscular atrophy.

Eugenio Mercuri1,2, Charlotte J Sumner3,4, Francesco Muntoni5,6, Basil T Darras7, Richard S Finkel8.   

Abstract

Spinal muscular atrophy (SMA) is a neurodegenerative disorder caused by mutations in SMN1 (encoding survival motor neuron protein (SMN)). Reduced expression of SMN leads to loss of α-motor neurons, severe muscle weakness and often early death. Standard-of-care recommendations for multidisciplinary supportive care of SMA were established in the past few decades. However, improved understanding of the pathogenetic mechanisms of SMA has led to the development of different therapeutic approaches. Three treatments that increase SMN expression by distinct molecular mechanisms, administration routes and tissue biodistributions have received regulatory approval with others in clinical development. The advent of the new therapies is redefining standards of care as in many countries most patients are treated with one of the new therapies, leading to the identification of emerging new phenotypes of SMA and a renewed characterization of demographics owing to improved patient survival.
© 2022. Springer Nature Limited.

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Year:  2022        PMID: 35927425     DOI: 10.1038/s41572-022-00380-8

Source DB:  PubMed          Journal:  Nat Rev Dis Primers        ISSN: 2056-676X            Impact factor:   65.038


  207 in total

1.  Genetic risk assessment in carrier testing for spinal muscular atrophy.

Authors:  Shuji Ogino; Debra G B Leonard; Hanna Rennert; Warren J Ewens; Robert B Wilson
Journal:  Am J Med Genet       Date:  2002-07-15

2.  Incidence, prevalence, and gene frequency studies of chronic childhood spinal muscular atrophy.

Authors:  J Pearn
Journal:  J Med Genet       Date:  1978-12       Impact factor: 6.318

3.  The implementation of newborn screening for spinal muscular atrophy: the Australian experience.

Authors:  Didu S T Kariyawasam; Jacqueline S Russell; Veronica Wiley; Ian E Alexander; Michelle A Farrar
Journal:  Genet Med       Date:  2019-10-14       Impact factor: 8.822

4.  Molecular analysis of spinal muscular atrophy and modification of the phenotype by SMN2.

Authors:  Matthew D Mailman; John W Heinz; Audrey C Papp; Pamela J Snyder; Mary S Sedra; Brunhilde Wirth; Arthur H M Burghes; Thomas W Prior
Journal:  Genet Med       Date:  2002 Jan-Feb       Impact factor: 8.822

5.  Pan-ethnic carrier screening and prenatal diagnosis for spinal muscular atrophy: clinical laboratory analysis of >72,400 specimens.

Authors:  Elaine A Sugarman; Narasimhan Nagan; Hui Zhu; Viatcheslav R Akmaev; Zhaoqing Zhou; Elizabeth M Rohlfs; Kerry Flynn; Brant C Hendrickson; Thomas Scholl; Deborah Alexa Sirko-Osadsa; Bernice A Allitto
Journal:  Eur J Hum Genet       Date:  2011-08-03       Impact factor: 4.246

6.  A multi-source approach to determine SMA incidence and research ready population.

Authors:  Ingrid E C Verhaart; Agata Robertson; Rebecca Leary; Grace McMacken; Kirsten König; Janbernd Kirschner; Cynthia C Jones; Suzanne F Cook; Hanns Lochmüller
Journal:  J Neurol       Date:  2017-06-20       Impact factor: 4.849

7.  Newborn screening for spinal muscular atrophy in Germany: clinical results after 2 years.

Authors:  Katharina Vill; Oliver Schwartz; Heike Kölbel; Wolfgang Müller-Felber; Astrid Blaschek; Dieter Gläser; Uta Nennstiel; Brunhilde Wirth; Siegfried Burggraf; Wulf Röschinger; Marc Becker; Ludwig Czibere; Jürgen Durner; Katja Eggermann; Bernhard Olgemöller; Erik Harms; Ulrike Schara
Journal:  Orphanet J Rare Dis       Date:  2021-03-31       Impact factor: 4.123

8.  Implementation of population-based newborn screening reveals low incidence of spinal muscular atrophy.

Authors:  Denise M Kay; Colleen F Stevens; April Parker; Carlos A Saavedra-Matiz; Virginia Sack; Wendy K Chung; Claudia A Chiriboga; Kristin Engelstad; Emma Laureta; Osman Farooq; Emma Ciafaloni; Bo Hoon Lee; Sohail Malek; Simona Treidler; Yaacov Anziska; Leslie Delfiner; Ai Sakonju; Michele Caggana
Journal:  Genet Med       Date:  2020-05-18       Impact factor: 8.822

Review 9.  Prevalence, incidence and carrier frequency of 5q-linked spinal muscular atrophy - a literature review.

Authors:  Ingrid E C Verhaart; Agata Robertson; Ian J Wilson; Annemieke Aartsma-Rus; Shona Cameron; Cynthia C Jones; Suzanne F Cook; Hanns Lochmüller
Journal:  Orphanet J Rare Dis       Date:  2017-07-04       Impact factor: 4.123

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