Congenital scoliosis: a narrative review and proposal of a treatment algorithm

Sebaaly, Amer; Daher, Mohammad; Salameh, Bendy; Ghoul, Ali; George, Samuel; Roukoz, Sami

doi:10.1530/eor-21-0121

Cited by 9 publications

(4 citation statements)

References 77 publications

(130 reference statements)

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“…To date, due to the unknown etiology and factors that influence CS, there has been no essential progress in the treatment of patients with CS, which mainly includes observation, bracing, and surgery. [ 20 ] However, although surgery is the only curative tool, it is very risky and costly. [ 3 , 21 ] Thus, it is important to reveal the pathology and mechanism of CS to explore better treatment options.…”

Section: Discussionmentioning

confidence: 99%

Promotion effect of FGF23 on osteopenia in congenital scoliosis through FGFr3/TNAP/OPN pathway

Zhang

Xiang

et al. 2023

Chinese Medical Journal

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Background: Congenital scoliosis (CS) is a complex spinal malformation of unknown etiology with abnormal bone metabolism. Fibroblast growth factor 23 (FGF23), secreted by osteoblasts and osteocytes, can inhibit bone formation and mineralization. This research aims to investigate the relationship between CS and FGF23. Methods: We collected peripheral blood from two pairs of identical twins for methylation sequencing of the target region. FGF23 mRNA levels in the peripheral blood of CS patients and age-matched controls were measured. Receiver operator characteristic (ROC) curve analyses were conducted to evaluate the specificity and sensitivity of FGF23. The expression levels of FGF23 and its downstream factors fibroblast growth factor receptor 3 (FGFr3)/tissue non-specific alkaline phosphatase (TNAP)/osteopontin (OPN) in primary osteoblasts from CS patients (CS-Ob) and controls (CT-Ob) were detected. In addition, the osteogenic abilities of FGF23-knockdown or FGF23-overexpressing Ob were examined. Results: DNA methylation of the FGF23 gene in CS patients was decreased compared to that of their identical twins, accompanied by increased mRNA levels. CS patients had increased peripheral blood FGF23 mRNA levels and decreased computed tomography (CT) values compared with controls. The FGF23 mRNA levels were negatively correlated with the CT value of the spine, and ROCs of FGF23 mRNA levels showed high sensitivity and specificity for CS. Additionally, significantly increased levels of FGF23, FGFr3, OPN, impaired osteogenic mineralization and lower TNAP levels were observed in CS-Ob. Moreover, FGF23 overexpression in CT-Ob increased FGFr3 and OPN levels and decreased TNAP levels, while FGF23 knockdown induced downregulation of FGFr3 and OPN but upregulation of TNAP in CS-Ob. Mineralization of CS-Ob was rescued after FGF23 knockdown. Conclusions: Our results suggested increased peripheral blood FGF23 levels, decreased bone mineral density in CS patients, and a good predictive ability of CS by peripheral blood FGF23 levels. FGF23 may contribute to osteopenia in CS patients through FGFr3/TNAP/OPN pathway.

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Section: Discussionmentioning

confidence: 99%

Promotion effect of FGF23 on osteopenia in congenital scoliosis through FGFr3/TNAP/OPN pathway

Zhang

Xiang

et al. 2023

Chinese Medical Journal

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“…CS is an early-onset spinal deformity often accompanied by other systemic malformations affecting the spinal cord, heart, kidney, and gastrointestinal system [2]. The adverse health effects of CS, such as impaired lung function, are typically more severe than those associated with IS, and over 70% of CS cases progress aggressively, requiring surgical intervention [52]. Therefore, although the prevalence of CS in this study is relatively lower, the disease burden caused by CS should not be overlooked.…”

Section: Estimated Prevalence and Cases Of Is Among Chinese Children ...mentioning

confidence: 99%

Variations in the prevalence of scoliosis by age, sex, geographic region, and subtype among Chinese children: A systematic review and modelling study

Cao,

Sun,

Zheng

et al. 2024

J Glob Health

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Background Scoliosis is a prevalent spinal condition typically detected in children, yet its prevalence in China has not been fully understood. This study aimed to estimate the prevalence of scoliosis among Chinese children and explore its variations by age, sex, geographic region, and subtype. MethodsWe conducted a comprehensive search of seven bibliographic databases to identify epidemiological studies that reported the prevalence of scoliosis among Chinese children published since 1990. Scoliosis was defined as a Cobb angle equal to or greater than 10°, as detected radiographically. We used a multilevel mixed-effect meta-regression to estimate the age-and sex-specific prevalence of idiopathic scoliosis (IS). The overall prevalence and total cases of IS among Chinese children aged 5-18 years in 2020 were generated by applying the China census data. A random-effects meta-analysis was conducted to estimate the pooled prevalence of IS with varying curvatures (10-19°, 20-39°, and ≥40°) and locations (thoracic curve, lumbar curve, thoracolumbar curve, and double curve), as well as congenital scoliosis (CS).Results A total of 46 articles covering 1 112 163 Chinese children were included. The prevalence of IS ranged from 0.06% (95% confidence interval (CI) = 0.00-0.26) among children aged five years to 1.44% (95% CI = 0.89-2.13) among those aged 18 years, peaking at 1.79% (95% CI = 1.18-2.53) among those aged 16 years. In 2020, the overall prevalence of IS among Chinese children aged 5-18 was 0.79% (95% CI = 0.45-1.27), translating to an estimated 1.81 million (95% CI = 1.04-2.91) affected children. Notably, IS prevalence was nearly double in girls compared to boys (1.03%, 95% CI = 0.63-1.57 vs. 0.58%, 95% CI = 0.29-1.01). Among the six geographic regions, the prevalence of IS was the highest in Northwest China (1.54%, 95% CI = 0.75-2.65) and the lowest in North China (0.33%, 95% CI = 0.11-0.73). The most commonly identified IS subtypes were curvatures of 10-19° and thoracolumbar curves, with a pooled prevalence of 7.14‰ (95% CI = 3.44-12.15) and 2.00‰ (95% CI = 0.83-3.65), respectively. As for CS, the pooled prevalence was 3.03 per 10 000 (95% CI = 1.88-4.43). ConclusionsThe prevalence of scoliosis among Chinese children exhibits considerable variations across age, sex, geographic regions, and subtypes. It is imperative to develop targeted public health strategies and optimise the allocation of regional health resources. High-quality epidemiological research is still needed to unmask the diverse burden of scoliosis in children.Registration PROSPERO CRD42023406260.

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“…Congenital scoliosis, which makes up to 10% of all scoliotic deformities, is defined as a spine deformity due to congenital vertebral malformation(s) [ 1 ]. A recent population-based study reported a total prevalence of 2.2 per 10,000 births [ 2 ], and pregestational diabetes appears to be a significant risk factor for these anomalies [ 3 ].…”

Section: Introductionmentioning

confidence: 99%

Hidden blood loss and bleeding characteristics in children with congenital scoliosis undergoing spinal osteotomies

Raitio,

Heiskanen,

Soini

et al. 2024

International Orthopaedics (SICOT)

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Purpose Spinal osteotomies are often essential in the treatment of congenital scoliosis. Risk factors for bleeding in these patients needing extracavitatory approaches, especially hidden blood loss, are sparsely investigated. We aimed to investigate the bleeding characteristics and hidden blood loss in paediatric patients undergoing spinal osteotomies for congenital scoliosis. Methods A retrospective analysis identified all patients with congenital scoliosis were retrospectively identified from the prospectively collected spine register from 2010 to 2022. Operative technique, perioperative laboratory results and imaging studies were extracted. The primary outcome was total blood loss including intraoperative, drain output and hidden blood loss. Results Fifty-seven children (32 boys) with a mean age of 8.3 years underwent spinal osteotomy for congenital scoliosis. Posterolateral hemivertebrectomy was sufficient in 34 (59%) patients, while vertebral column resection (VCR) was required in 23 patients. Total bleeding averaged 792 (523) ml accounting for 42% of the estimated blood volume. Hidden blood loss accounted for 40% of total bleeding and 21% of estimated blood volume with a mean of 317 (256) ml. VCR was associated with greater intraoperative and total bleeding than hemivertebrectomies (p = 0.001 and 0.007, respectively). After adjusting for patient weight and fusion levels, hidden blood loss was larger in hemivertebrectomies (4.18 vs. 1.77 ml/kg/fused level, p = 0.049). In multivariable analysis, intraoperative blood loss was inversely correlated with preoperative erythrocyte levels. Younger age was associated with significantly greater drain, hidden and total blood loss. Conclusion Hidden blood loss constitutes a significant portion (40%) of total bleeding in congenital scoliosis surgery. Younger age is a risk factor for bleeding and the hidden blood loss should be taken into consideration in their perioperative management.

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Congenital scoliosis: a narrative review and proposal of a treatment algorithm

Cited by 9 publications

References 77 publications

Promotion effect of FGF23 on osteopenia in congenital scoliosis through FGFr3/TNAP/OPN pathway

Promotion effect of FGF23 on osteopenia in congenital scoliosis through FGFr3/TNAP/OPN pathway

Variations in the prevalence of scoliosis by age, sex, geographic region, and subtype among Chinese children: A systematic review and modelling study

Hidden blood loss and bleeding characteristics in children with congenital scoliosis undergoing spinal osteotomies

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