Retrospective Cohort Study
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Orthop. Jun 18, 2023; 14(6): 411-426
Published online Jun 18, 2023. doi: 10.5312/wjo.v14.i6.411
Surgical and long-term functional outcomes of patients with Duchenne muscular dystrophy following spinal deformity correction
Simon Roberts, Ayesha Arshad, Athanasios I Tsirikos
Simon Roberts, Department of Spinal Surgery, Leeds Teaching Hospitals NHS Trust, Leeds LS1 3EX, United Kingdom
Ayesha Arshad, Athanasios I Tsirikos, Scottish National Spine Deformity Centre, Royal Hospital for Children and Young People, Edinburgh EH16 4TJ, United Kingdom
Author contributions: Tsirikos AI was the guarantor, treated the patients, designed the study, participated in acquisition, analysis and interpretation of the data and drafted the manuscript; Arshad A and Roberts S participated in analysis and interpretation of the data and drafted the manuscript.
Institutional review board statement: This retrospective cohort study does not require ethical review.
Informed consent statement: The requirement for a signed informed consent was waived.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
Data sharing statement: No additional data are available.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Athanasios I Tsirikos, FRCS, MD, PhD, Senior Lecturer, Surgeon, Scottish National Spine Deformity Centre, Royal Hospital for Children and Young People, 50 Little France Crescent, Edinburgh Bioquarter, Edinburgh EH16 4TJ, United Kingdom. thanos.tsirikos@nhslothian.scot.nhs.uk
Received: December 27, 2022
Peer-review started: December 27, 2022
First decision: March 14, 2023
Revised: March 29, 2023
Accepted: May 15, 2023
Article in press: May 15, 2023
Published online: June 18, 2023
Processing time: 173 Days and 14.7 Hours
Abstract
BACKGROUND

Life expectancy in patients with Duchenne muscular dystrophy (DMD) has improved due to advances in medical care. DMD patients develop progressive spinal deformity after loss of ambulatory function and onset of wheelchair dependence for mobility. There is limited published data on the effect of spinal deformity correction on long-term functional outcomes, quality of life (QoL), and satisfaction in DMD patients.

AIM

To investigate the long-term functional outcomes following spinal deformity correction in DMD patients.

METHODS

This was a retrospective cohort study from 2000-2022. Data was collected from hospital records and radiographs. At follow-up, patients completed the muscular dystrophy spine questionnaire (MDSQ). Statistical analysis was performed by linear regression analysis and ANOVA to analyse clinical and radiographic factors significantly associated with MDSQ scores.

RESULTS

Forty-three patients were included with mean age 14.4 years at surgery. Spino-pelvic fusion was performed in 41.9% of patients. Mean surgical time was 352.1 min and mean blood loss was 36% of estimated total blood volume. Mean hospital stay was 14.1 d. Postoperative complications occurred in 25.6% of patients. Mean preoperative scoliosis was 58°, pelvic obliquity 16.4°, thoracic kyphosis 55.8°, lumbar lordosis 11.1°, coronal balance 3.8 cm, and sagittal balance + 6.1 cm. Mean surgical correction of scoliosis was 79.2% and of pelvic obliquity was 80.8%. Mean follow-up was 10.9 years (range: 2-22.5). Twenty-four patients had died at follow-up. Sixteen patients completed the MDSQ at mean age 25.4 years (range 15.2-37.3). Two patients were bed-ridden and 7 were on ventilatory support. Mean MDSQ total score was 38.1. All 16 patients were satisfied with the results of spinal surgery and would choose surgery again if offered. Most patients (87.5%) reported no severe back pain at follow-up. Factors significantly associated with functional outcomes (MDSQ total score) included greater duration of post-operative follow-up, age, scoliosis postoperatively, correction of scoliosis, increased lumbar lordosis postoperatively, and greater age at loss of independent ambulation.

CONCLUSION

Spinal deformity correction in DMD patients leads to positive long-term effects on QoL and high patient satisfaction. These results support spinal deformity correction to improve long-term QoL in DMD patients.

Keywords: Duchenne muscular dystrophy; Scoliosis; Surgical; Functional; Outcomes

Core Tip: Duchenne muscular dystrophy (DMD) patients develop progressive spinal deformity after loss of independent ambulation. There is limited data on the effect of spinal deformity correction on long-term functional outcomes or satisfaction in DMD patients. This retrospective cohort study investigated long-term functional outcomes following spinal deformity correction in DMD patients, reporting clinical, surgical, radiographic and functional outcomes. All patients were satisfied with surgical outcomes at long-term follow-up. Surgical correction of spinal deformity can have favourable long-term effects on quality of life (QoL) in DMD patients. These results support surgical correction of spinal deformity to improve long-term QoL in DMD patients with spinal deformity.