Literature DB >> 31243392

AAV9 Vector: a Novel modality in gene therapy for spinal muscular atrophy.

Rithu Pattali1, Yongchao Mou1,2, Xue-Jun Li3,4.   

Abstract

Spinal muscular atrophy (SMA), the leading genetic cause of infant mortality, is characterized by the deterioration of alpha motor neurons in the brainstem and spinal cord. Currently, there is no cure for SMA, which calls for an urgent need to explore affordable and effective therapies and to maximize patients' independence and quality of life. Adeno-associated virus (AAV) vector, one of the most promising and well-investigated vehicles for delivering transgenes, is a compelling candidate for gene therapy. Some of the hallmarks of AAVs are their nonpathogenicity, inability to incur an immune response, potential to achieve robust transgene expression, and varied tropism for several tissues of the body. Recently, these features were harnessed in a clinical trial conducted by AveXis in SMA patients, where AAV9 was employed as a vehicle for one-time administration of the SMN gene, the causative gene in SMA. The trial demonstrated remarkable improvements in motor milestones and rates of survival in the patients. This review focuses on the advent of SMA gene therapy and summarizes different preclinical studies that were conducted leading up to the AAV9-SMA trial in SMA patients.

Entities:  

Year:  2019        PMID: 31243392     DOI: 10.1038/s41434-019-0085-4

Source DB:  PubMed          Journal:  Gene Ther        ISSN: 0969-7128            Impact factor:   5.250


  20 in total

1.  Effects of Altering HSPG Binding and Capsid Hydrophilicity on Retinal Transduction by AAV.

Authors:  Sean M Crosson; Antonette Bennett; Diego Fajardo; James J Peterson; Hangning Zhang; Wei Li; Matthew T Leahy; Colin K Jennings; Ryan F Boyd; Sanford L Boye; Mavis Agbandge-McKenna; Shannon E Boye
Journal:  J Virol       Date:  2021-03-03       Impact factor: 5.103

Review 2.  Evaluating the state of the science for adeno-associated virus integration: An integrated perspective.

Authors:  Denise E Sabatino; Frederic D Bushman; Randy J Chandler; Ronald G Crystal; Beverly L Davidson; Ricardo Dolmetsch; Kevin C Eggan; Guangping Gao; Irene Gil-Farina; Mark A Kay; Douglas M McCarty; Eugenio Montini; Adora Ndu; Jing Yuan
Journal:  Mol Ther       Date:  2022-06-10       Impact factor: 12.910

3.  Motor unit recovery following Smn restoration in mouse models of spinal muscular atrophy.

Authors:  Laura H Comley; Rachel A Kline; Alison K Thomson; Victoria Woschitz; Eric Villalón Landeros; Erkan Y Osman; Christian L Lorson; Lyndsay M Murray
Journal:  Hum Mol Genet       Date:  2022-09-10       Impact factor: 5.121

4.  Suppression of heterotopic ossification in fibrodysplasia ossificans progressiva using AAV gene delivery.

Authors:  Yeon-Suk Yang; Jung-Min Kim; Jun Xie; Sachin Chaugule; Chujiao Lin; Hong Ma; Edward Hsiao; Jaehyoung Hong; Hyonho Chun; Eileen M Shore; Frederick S Kaplan; Guangping Gao; Jae-Hyuck Shim
Journal:  Nat Commun       Date:  2022-10-19       Impact factor: 17.694

5.  Adeno-Associated Virus-Mediated Gene Therapy in the Mashlool, Atp1a3Mashl/+, Mouse Model of Alternating Hemiplegia of Childhood.

Authors:  Arsen S Hunanyan; Boris Kantor; Ram S Puranam; Courtney Elliott; Angela McCall; Justin Dhindsa; Promila Pagadala; Keri Wallace; Jordan Poe; Talha Gunduz; Aravind Asokan; Dwight D Koeberl; Mai K ElMallah; Mohamad A Mikati
Journal:  Hum Gene Ther       Date:  2021-02-12       Impact factor: 5.695

6.  Designer Receptors Exclusively Activated by Designer Drugs Approach to Treatment of Sleep-disordered Breathing.

Authors:  Thomaz Fleury Curado; Huy Pho; Carla Freire; Mateus R Amorim; Jordi Bonaventura; Lenise J Kim; Rachel Lee; Meaghan E Cabassa; Stone R Streeter; Luiz G Branco; Luiz U Sennes; Kenneth Fishbein; Richard G Spencer; Alan R Schwartz; Michael J Brennick; Michael Michaelides; David D Fuller; Vsevolod Y Polotsky
Journal:  Am J Respir Crit Care Med       Date:  2021-01-01       Impact factor: 21.405

7.  Adeno-associated Virus 9 Structural Rearrangements Induced by Endosomal Trafficking pH and Glycan Attachment.

Authors:  Judit J Penzes; Paul Chipman; Nilakshee Bhattacharya; Allison Zeher; Rick Huang; Robert McKenna; Mavis Agbandje-McKenna
Journal:  J Virol       Date:  2021-07-14       Impact factor: 5.103

Review 8.  Planet of the AAVs: The Spinal Cord Injury Episode.

Authors:  Katerina Stepankova; Pavla Jendelova; Lucia Machova Urdzikova
Journal:  Biomedicines       Date:  2021-05-28

9.  AAV9 gene transfer of cMyBPC N-terminal domains ameliorates cardiomyopathy in cMyBPC-deficient mice.

Authors:  Jiayang Li; Ranganath Mamidi; Chang Yoon Doh; Joshua B Holmes; Nikhil Bharambe; Rajesh Ramachandran; Julian E Stelzer
Journal:  JCI Insight       Date:  2020-09-03

Review 10.  One drug to treat many diseases: unlocking the economic trap of rare diseases.

Authors:  Karolina Pierzynowska; Teresa Kamińska; Grzegorz Węgrzyn
Journal:  Metab Brain Dis       Date:  2020-09-14       Impact factor: 3.584

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