Pediatric thoracic outlet syndrome: a systematic review and meta-analysis

Author:

Price Anthony12,Fredricks Nathan12,Truong Nina12,North Robert Y.3

Affiliation:

1. John Sealy School of Medicine, The University of Texas Medical Branch at Galveston, Texas;

2. Department of Neurosurgery, The University of Texas Medical Branch at Galveston, Texas;

3. Department of Neurosurgery, The University of Texas MD Anderson Cancer Center, Houston, Texas

Abstract

OBJECTIVE Thoracic outlet syndrome (TOS) is a complex disorder affecting the neurovascular structures of the upper extremity as they traverse from the neck and thorax to the upper extremity. This systematic review and meta-analysis focuses on pediatric TOS, offering insights into its clinical presentation, etiology, treatment modalities, and outcomes in contrast to those reported in adult TOS. METHODS A comprehensive search for pediatric TOS in the PubMed database using PRISMA guidelines identified 6 relevant studies published between 2008 and 2022. In total, 227 pediatric TOS cases in 216 patients were analyzed. Data categories explored for TOS in pediatric patients included study design, number of patients included, mean age and sex of patients, TOS type, laterality, bony abnormalities, time to surgery, symptoms, treatment modalities, initial surgical technique, surgical complications, percent lost to follow-up, mean follow-up period, and treatment outcome. RESULTS The results from the 6 studies of 216 patients show a distinct pattern in pediatric TOS, with a 1.84:1 female-to-male ratio, a mean age of 15.49 years, and a lower prevalence of neurogenic TOS (75%, 95% CI 0.41–0.93; I2 = 86%, p < 0.01) compared with the prevailing literature on adults (87.5%–99%). Venous and arterial TOS accounted for a higher proportion of cases in pediatric patients than in adults, challenging the traditional adult-oriented perspective. Right-sided presentations were more common, reflecting right-arm dominance in most individuals. Additionally, bony abnormalities were more common in adults (30%) than in children (10.65%). Treatments involved mixed methods, predominantly using combinations of muscle resection (95.26%), neurolysis (78.02%), and bone resection (72.41%). Patients had high rates of symptom improvement (89%, 95% CI 0.67–0.97; I2 = 85%, p < 0.01) following surgery, with improvement of symptoms ranging from slight to complete relief. Complications were infrequent (5.66%), and most patients reported positive outcomes. The limitations of this analysis include subjective diagnostic and reporting criteria for TOS given its broad range of presentations. CONCLUSIONS This systematic review and meta-analysis brings to light the distinctive characteristics of pediatric TOS and underscores the importance of recognizing these differences to ensure accurate diagnosis and effective treatment in this patient population. Further research is needed to understand the predictive value of conservative treatments, especially in pediatric TOS cases.

Publisher

Journal of Neurosurgery Publishing Group (JNSPG)

Reference23 articles.

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2. Outcomes following operative management of thoracic outlet syndrome in the pediatric patients;Matos JM,2018

3. Managing pediatric thoracic outlet syndrome;Rehemutula A,2015

4. Thoracic outlet syndrome with right subclavian artery dilatation in a child - transaxillary resection of the pediatric cervical rib;Şen S,2007

5. Subclavian artery aneurysm caused by cervical rib: case report and review;Engel A,1989

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