Literature DB >> 32809150

Impact of growth friendly interventions on spine and pulmonary outcomes of patients with spinal muscular atrophy.

Ishaan Swarup1,2, Elle M MacAlpine1, Oscar H Mayer3, Robert K Lark4, John T Smith5, Michael G Vitale6, John M Flynn1, Jason B Anari1, Patrick J Cahill7.   

Abstract

PURPOSE: Patients with spinal muscular atrophy (SMA) are often treated with growth friendly devices such as vertical expandable prosthetic titanium rib(VEPTR) and magnetically controlled growing rods(MCGR) to correct spinal deformity and improve pulmonary function. There is limited data on this topic, and the purpose of this study was to assess the effect of these constructs and the addition of chest wall support (CWS) on spinal deformity, thorax morphology and pulmonary outcomes.
METHODS: This is a retrospective analysis of prospectively collected data. We included patients with chest wall deformity and scoliosis secondary to SMA who were treated with growth friendly interventions and had two-year follow-up. Descriptive statistics and univariate analyses were performed.
RESULTS: This study included 66 patients (25% MCGR, 73% VEPTR, 2% unknown). Approximately 23% of constructs included CWS. The average Cobb angle improved from 67° (SD: 27°) to 50° (SD: 26°) at 2 years in patients with CWS (p = 0.02), and from 59° (SD: 20°) to 46° (SD: 15°) at 2 years in patients without CWS (p < 0.01). Hemithorax height improved in patients treated with and without CWS (p = 0.01), but hemithorax width only improved in patients with CWS (p = 0.01). One patient with CWS and two patients without CWS required additional respiratory support at 2 years. The rates of postoperative complications were not significantly different in patients treated with and without CWS (p = 0.31).
CONCLUSIONS: Growth friendly constructs improve spinal deformity and may be effective in altering the progression toward respiratory failure in patients with SMA. Patients treated with CWS have significant improvements in thorax morphology compared to patients without CWS.

Entities:  

Keywords:  Early-onset scoliosis; Growing rod; Spinal muscular atrophy; Thoracic insufficiency syndrome; Vertical expandable prosthetic titanium rib

Mesh:

Substances:

Year:  2020        PMID: 32809150     DOI: 10.1007/s00586-020-06564-8

Source DB:  PubMed          Journal:  Eur Spine J        ISSN: 0940-6719            Impact factor:   3.134


  4 in total

Review 1.  Scoliosis and the impact in neuromuscular disease.

Authors:  Oscar Henry Mayer
Journal:  Paediatr Respir Rev       Date:  2014-11-05       Impact factor: 2.726

2.  Effects of magnetically controlled growing rods surgery on pulmonary function in young subjects with spinal muscular atrophy type 2 and other neuromuscular scoliosis.

Authors:  Luca Colombo; Carlotta Martini; Chiara Bersanini; Francesca Izzo; Jorge H Villafañe; Pedro Berjano; Claudio Lamartina
Journal:  J Neurosurg Sci       Date:  2017-05-31       Impact factor: 2.279

Review 3.  Antisense oligonucleotides in neurological disorders.

Authors:  Claudia D Wurster; Albert C Ludolph
Journal:  Ther Adv Neurol Disord       Date:  2018-05-23       Impact factor: 6.570

Review 4.  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

  4 in total
  3 in total

1.  Surgical treatment of spinal deformities in spinal muscular atrophy: a single-center experience from China.

Authors:  Zhen Wang; Erwei Feng; Yang Jiao; Jiachen Lin; Junduo Zhao; Weiyun Chen; Jianxiong Shen
Journal:  Eur Spine J       Date:  2022-08-16       Impact factor: 2.721

2.  Scoliosis Orthopedic Surgery Combined With Nusinersen Intrathecal Injection Significantly Improved the Outcome of Spinal Muscular Atrophy Patient: A Case Report.

Authors:  Beiyu Xu; Cuijie Wei; Xiao Hu; Wenzhu Li; Zhen Huang; Chengli Que; Jianxing Qiu; Chunde Li; Hui Xiong
Journal:  Front Neurol       Date:  2022-04-25       Impact factor: 4.086

Review 3.  Thoracic insufficiency syndrome: Approaches to assessment and management.

Authors:  Katharine Tsukahara; Oscar Henry Mayer
Journal:  Paediatr Respir Rev       Date:  2022-03-07       Impact factor: 5.526

  3 in total

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