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Journal of Peking University(Health Sciences) >

2025 , Vol. 57 >Issue 5: 941 - 946

DOI: https://doi.org/10.19723/j.issn.1671-167X.2025.05.019

Establishment and validation of a risk prediction model for scoliosis after Nuss procedure in children and young adults with pectus excavatum

  • Bowen LI ,
  • Qiang ZHANG , * ,
  • Yixin SUN
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  • Department of Thoracic Surgery, Beijing Jishuitan Hospital Affiliated to Capital Medical University, National Center for Orthopedics, Beijing 100035, China
ZHANG Qiang, e-mail:

Received date: 2024-06-16

  Online published: 2025-09-04

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Abstract

Objective: To identify the risk factors associated with the development of post-Nuss procedure scoliosis in pectus excavatum patients and to establish a relevant predictive model. Methods: A retro-spective review was conducted on pectus excavatum patients who underwent Nuss procedure in Department of Thoracic Surgery at Beijing Jishuitan Hospital between January 2018 and February 2023. We gathered the patient demographic information (including age, sex, height, weight, and body mass index) and diagnostic imaging results (echocardiogram, chest CT scan, full-spine radiography, and PA/lateral chest X-rays), and measurements of Haller index, asymmetry index, sternal torsion angle (STA) index, and Cobb angle changes. The risk of post-Nuss procedure scoliosis was assessed. Cox regression analysis was performed to identify independent risk factors for scoliosis development in the pectus excavatum patients. Based on the results of the Cox regression analysis, a risk prediction model was established, and its specific predictive performance was assessed through internal cross-validation. Results: A total of 59 pectus excavatum patients who underwent Nuss procedure were included after applying inclusion and exclusion criteria. The median follow-up duration was 6.84 months, and the follow-up results showed significant improvements in Haller index, STA index, and asymmetry index postoperatively. Twelve patients developed scoliosis 3 months after Nuss procedure, while 47 patients did not, the incidence of scoliosis was 20.3%. Cox regression analysis identified preoperative pulmonary artery hypertension and preoperative asymmetry index as independent risk factors for post-Nuss procedure scoliosis. A predictive model was constructed based on single-factor Cox regression analysis results, incorporating age, height, weight, body mass index, preoperative pulmonary artery hypertension, preoperative Haller index, STA index, asymmetry index, and Cobb angle. The receiver operating characteristic (ROC) curve was plotted, and the area under the curve (AUC) for the overall predictive model was calculated to be 0.995. A calibration curve demonstrated good alignment between predicted values and actual values. Conclusion: Nuss procedure achieved favorable clinical outcomes. However, postoperative scoliosis emerged as a significant complication with a high incidence rate. Pulmonary artery hypertension and asymmetry index were independent predictors of post-Nuss procedure scoliosis. The predictive model developed in this study demonstrated robust performance in estimating the risk of postoperative scoliosis.

Key words： Pectus excavatum; Nuss surgery; Scoliosis; Risk factors; Nomograms

Cite this article

Bowen LI , Qiang ZHANG , Yixin SUN . Establishment and validation of a risk prediction model for scoliosis after Nuss procedure in children and young adults with pectus excavatum[J]. Journal of Peking University(Health Sciences), 2025 , 57(5) : 941 -946 . DOI: 10.19723/j.issn.1671-167X.2025.05.019

漏斗胸是一种常见的先天性胸廓畸形，其特征为胸骨向内凹陷形成漏斗状，导致胸廓畸形和心肺功能受损[1-2]。Nuss手术作为一种微创胸廓矫正技术被引入，并逐渐成为漏斗胸治疗的首选方法之一[1, 3]。相较于传统的开放性手术，Nuss手术具有创伤小、康复快、并发症少等优势[4-5]。尽管Nuss手术在改善漏斗胸患者胸廓形态和生活质量方面表现出色，但其术后可能导致脊柱侧弯的发生，影响患者预后[6-7]。
脊柱侧弯是一种常见的脊柱畸形，其特征为脊柱在前后方向呈S形弯曲，导致脊柱的不稳定性和不良姿态[8]。Nuss手术后脊柱侧弯的发生可能与胸廓矫正后的生物力学改变有关[9]，漏斗胸患者自身胸廓畸形已导致脊柱受到异常的应力[10]，Nuss手术通过放置金属支架来矫正胸廓形态，但这种突然的形态改变可能进一步扰乱胸廓-脊柱-肋骨-肌肉系统的生物力学平衡，引起脊柱侧弯[11]。Nuss手术矫正胸廓后，胸廓前后径增加，横径减小，这可能导致肋骨和胸壁肌肉的附着点发生改变，进而改变施加在脊柱上的张力和压力，特别是在不对称性胸廓畸形患者中更为明显[12]。因此，了解Nuss手术后脊柱侧弯的发生风险及其影响因素，对于预防和管理该并发症具有重要意义[13]。
本研究通过收集临床数据和影像学资料，旨在建立针对Nuss术后脊柱侧弯风险的预测模型，并验证模型的准确性和可靠性，期望能为临床医生提供一个科学有效的工具，辅助术前风险评估和手术决策，降低术后脊柱侧弯的发生率，进一步提高Nuss手术的疗效。

1 资料与方法

1.1 研究对象

回顾性选取2018年1月至2023年2月于北京积水潭医院胸外科行Nuss手术治疗的漏斗胸患者。纳入标准: (1)年龄10~30岁；(2)没有重大手术并发症，未合并其他先天性畸形或疾病；(3)CT检查提示Haller指数>3.20；(4)术前无脊柱侧弯。排除标准：(1)临床资料不完整，缺乏影像学数据；(2)因外伤或先天性心脏病手术等导致前胸壁凹陷；(3)接受过传统有创手术。
收集患者的以下资料：(1)人口学资料，包括年龄、性别、身高、体重、体重指数；(2)心功能指标，术前超声心动图是否报告存在二尖瓣脱垂、二尖瓣/主动脉瓣关闭不全、三尖瓣关闭不全、肺动脉高压；(3)影像学资料，患者术后3个月时的CT或X线检查结果。

1.2 影像学指标

1.2.1 Haller指数

Haller指数是目前国际上普遍采用的判断漏斗胸畸形严重程度的指数，其为同一张影像片上的胸腔最大横径与最小前后径的比值。本研究利用胸部CT图像计算Haller指数，将Haller指数2.0~3.2定义为轻度漏斗胸，3.2~3.5定义为中度漏斗胸，>3.5定义为重度漏斗胸。

1.2.2 不对称指数

不对称指数(asymmetry index)是评价漏斗胸胸廓对称情况的指标，其值为同一张影像片上左半胸与右半胸最大前后径的比值减1。本研究利用CT图像计算胸部不对称指数，健康人在-0.05~ +0.05之间，低于或高于此范围均表示胸廓不对称。

1.2.3 Cobb角

Cobb角是评估脊柱侧弯严重程度的金标准，通过站立位全脊柱X线片进行测量。测量步骤如下：(1)确定侧弯的上、下端椎，即倾斜最严重的椎体，其椎间隙一侧最宽。(2)画参考线：沿上端椎的上终板(upper endplate)和下端椎的下终板(lower endplate)各画一条直线。(3)计算夹角：若两线相交，直接测量交角；若两线不相交，则作垂线后测量垂线夹角。脊柱侧弯的治疗方案根据Cobb角大小确定：＜25°时继续观察，25°~45°时佩戴脊柱支具治疗，＞45°时行手术治疗。

1.2.4 胸骨扭转角指数

本研究使用胸部CT图像计算胸骨扭转角(sternal torsion angle，STA)指数。于胸部CT矢状位重建图像上确定胸骨后凹最深点，并在该点处作一条垂直于前胸壁的直线，测量该直线的长度，记为a；然后，在同一层面上测量胸廓前后径，即胸骨后凹最深点至脊柱前缘的距离，记为b。STA指数的计算公式为：STA指数=a/b。

1.3 统计学方法

采用SPSS 24.0完成统计分析。符合正态分布的计量资料用均数±标准差表示，组间比较采用t检验；不符合正态分布的计量资料采用中位数(P25，P75)表示，组间比较采用Wilcoxon秩和检验；计数资料用例数(百分比)表示。采用R 4.1.0中的survi-val、rms、glmnet、randomForestSRC、timeROC软件包进行多因素Cox比例风险回归，建立预测模型并对其进行验证。P < 0.05为差异有统计学意义。

2 结果

2.1 漏斗胸患者Nuss术前基本情况

本研究共纳入59例行Nuss手术治疗漏斗胸的患者，其中男性52例，女性7例，患者的基本情况详见表 1。
表1 漏斗胸患者Nuss术前基本情况(n=59)

Table 1 Baseline characteristics of pectus excavatum patients before Nuss procedure (n=59)

Characteristics Values
Age/years, ${\bar x}$±s 16.0±3.99
Gender, n(%)
  Male 52 (88.1)
  Female 7 (11.9)
Height/cm, ${\bar x}$±s 134±8.71
Weight/kg, ${\bar x}$±s 38.3±8.15
Body mass index/(kg/m2), ${\bar x}$±s 20.0±2.36
Mitral valve prolapse, n(%) 3 (5.08)
Mitral regurgitation, n(%) 6 (10.2)
Tricuspid regurgitation, n(%) 1 (1.69)
Pulmonary artery hypertension, n(%) 4 (6.78)

2.2 漏斗胸患者Nuss术后随访数据

患者的中位随访时间为6.84个月(4.5~12.0个月)。与术前相比，术后3个月Haller指数、STA指数、不对称指数显著改善，Cobb角明显增大(P < 0.05，表 2)。术后3个月共12例(20.3%)患者发生脊柱侧弯，均为单胸弯，其中5例凸向左侧，7例凸向右侧；术后3个月Cobb角范围为11°~16°，均予以随访观察。本组无发生双胸弯、胸腰弯和腰弯的患者。
表2 漏斗胸患者Nuss手术前后影像学指标变化

Table 2 Radiographic parameter changes before and 3 months after Nuss procedure in pectus excavatum patients

Radiographic parameters Before Nuss 3 months after Nuss P
Haller index, median (P25, P75) 3.20 (0.47, 5.35) 1.32 (0.18, 2.53) < 0.000 1
Sternal torsion angle index, median (P25, P75) 16.0 (5.75, 29.8) 11.8 (1.78, 21.6) < 0.000 1
Asymmetry index, median (P25, P75) 0.59 (0.33, 0.89) 0.32 (0.12, 0.48) < 0.000 1
Cobb angel, ${\bar x}$±s 7.16±2.45 8.86±3.20 0.001 6

2.3 Nuss术后脊柱侧弯的Cox比例风险回归分析

单因素Cox回归分析结果显示，年龄、身高、体重、体重指数及术前肺动脉压力、Haller指数、STA指数、不对称指数、Cobb角是漏斗胸患者Nuss术后脊柱侧弯的潜在风险因素(P < 0.05，表 3)。进一步的多因素Cox回归分析表明，术前肺动脉高压和不对称指数高是漏斗胸患者Nuss术后脊柱侧弯的独立危险因素(P < 0.05，表 4，图 1A和B)。
表3 漏斗胸患者Nuss术后合并脊柱侧弯的单因素Cox回归分析

Table 3 Univariate Cox regression analysis of factors associated with scoliosis in pectus excavatum patients after the Nuss procedure

Variable HR (95%CI) β SE P
Age 1.19 (1.05-1.36) 0.18 0.07 0.010
Gender 0.00 (0.00-Infinity) -18.20 6 900.00 1.000
Height 1.07 (1.01-1.14) 0.07 0.03 0.040
Weight 1.10 (1.02-1.19) 0.10 0.04 0.020
BMI 0.67 (0.49-0.90) -0.40 0.15 0.010
Mitral valve prolapse 4.75 (0.54-41.64) 1.56 1.11 0.160
Mitral regurgitation 1.54 (0.34-7.04) 0.43 0.78 0.580
Pulmonary artery hypertension 17.08 (4.66-62.62) 2.84 0.66 < 0.001
Haller index 0.25 (0.14-0.43) -1.40 0.29 < 0.001
STA index 0.68 (0.58-0.80) -0.39 0.08 < 0.001
Asymmetry index 0.00 (0.00-0.00) -19.70 4.30 < 0.001
Cobb angle 0.41 (0.28-0.61) -0.89 0.20 < 0.001

HR, hazard ratio; CI, confidence interval; β, regression coefficient; SE, standard error; BMI, body mass index; STA, sternal torsion angle.

表4 漏斗胸患者Nuss术后脊柱侧弯的多因素Cox回归分析

Table 4 Multivariate Cox regression analysis of factors associated with scoliosis in pectus excavatum patients after the Nuss procedure

Variable HR (95%CI) β SE P
Age 1.19 (0.96-1.48) 0.176 0.110 0.111
Height 1.09 (0.94-1.25) 0.083 0.073 0.259
Weight 1.04 (0.87-1.25) 0.040 0.092 0.664
BMI 1.13 (0.61-2.08) 0.119 0.313 0.704
Pulmonary artery hypertension 20.80 (1.37-314.00) 3.030 1.390 0.029
Haller index 39.9 (0.19-8 380) 3.690 2.730 0.177
STA index 0.47 (0.06-3.55) -0.749 1.030 0.466
Asymmetry index 1.8×10-54 (3.6×10-96-9.1×10-13) -124.000 49.000 0.011
Cobb angle 54.4 (0.31-9 650) 4.000 2.640 0.131

Abbreviations as in Table 3.

图1 漏斗胸Nuss术后合并脊柱侧弯患者风险预测模型的建立

Figure 1 Establishment of a risk prediction model for scoliosis in pectus excavatum patients after the Nuss procedure

A, B, forest plots showing the results of the multivariate Cox regression analysis, indicating that AI and PAH are key risk factors influencing postoperative scoliosis in patients; C, nomogram displaying the prediction model. BMI, body mass index; PAH, pulmonary arterial hypertension; STA, sternal torsion angle; AI, asymmetry index; Pr, prediction.

2.4 Nuss术后发生脊柱侧弯的风险预测模型的建立及验证

基于多因素Cox回归分析结果绘制列线图，建立Nuss术后发生脊柱侧弯的风险预测模型(图 1C)。对预测模型进行内部交叉验证，绘制3、6、12个月的时间依赖性受试者工作特征(receiver opera-ting characteristic，ROC)曲线，结果显示整体预测模型12个月时的曲线下面积为0.995，提示预测模型具有较好的区分度(图 2A)；绘制术后3、6、12个月的校准曲线，结果显示预测值与实际值重合度良好(图 2B~D)。
图2 漏斗胸Nuss术后合并脊柱侧弯患者风险预测模型的验证

Figure 2 Validation of the risk prediction model for scoliosis in pectus excavatum patients after the Nuss procedure

A, time-dependent receiver operating characteristic (ROC) curve at 3, 6, and 12 months evaluating the predictive performance of the overall prediction model on patient prognosis; B-D, calibration curves at 3, 6, and 12 months assessing the consistency between predicted and actual values respectively. AUC, area under the curve.

3 讨论

Nuss手术能够有效改善漏斗胸患者的Haller指数、STA指数和不对称指数，这些指标的改善意味着患者的心肺功能得到了改善，呼吸困难等临床症状可能得到明显缓解，有助于提高患者的生活质量[14-15]。Nuss手术具有微创性，相比于传统开放手术，术后疼痛感较轻，康复时间较短，患者能够更早地恢复正常日常生活，这也是其受到患者和医生欢迎的重要原因之一[16]。但Nuss术后脊柱侧弯的发生率较高，本组患者为20.3%。Nuss术后脊柱侧弯发生会引起疼痛加重、生活质量降低，严重时可能需要进行额外的矫正手术，给患者预后造成不良影响。因此，有效识别Nuss术后可能发生脊柱侧弯的高风险人群，具有重要的意义及价值。
本研究发现术前肺动脉高压是影响Nuss术后脊柱侧弯的独立危险因素之一。漏斗胸患者常伴随有心肺功能的损害，研究表明，漏斗胸患者的肺动脉收缩压和肺动脉舒张压均显著高于正常对照组，提示漏斗胸可能导致肺动脉高压。此外，还有研究发现，漏斗胸患者的平均肺动脉压力显著高于正常人群[17-18]。肺动脉高压导致Nuss术后脊柱侧弯发生的风险机制可能为：(1)肺动脉高压使右心室后负荷增加，进而引起右心室扩大和右心功能不全[19]，从而导致术后胸廓在矫正过程中无法得到充分的扩张，增加脊柱侧弯的风险；(2)肺动脉高压可引起肺血管重构，导致肺顺应性降低[20]，从而限制术后胸廓的扩张，增加脊柱侧弯的风险；(3)肺动脉高压还会导致呼吸功能受损，引起低氧血症和高碳酸血症[21]，对术后脊柱的正常发育产生不利影响，增加侧弯的风险。因此，术前需要对患者进行详细的心肺功能评估，确保肺动脉压力在可接受范围内，或者在手术前进行相应的调整和干预[22]。
不对称指数是通过计算胸部CT图像中左半胸和右半胸的前后径比值来评估胸廓对称性的指标，健康人群该数值通常在-0.05~+0.05之间，低于或高于此范围则表示胸廓不对称。因此，术前不对称指数的升高可能反映了患者术前的胸廓不对称性，胸廓不对称可能在Nuss手术后引发脊柱侧弯。既往研究结果也显示，胸廓不对称指数与Cobb角的相关系数为0.864，证实了胸廓不对称与Cobb角密切相关[23]。术前不对称指数升高可能通过以下机制增加Nuss术后脊柱侧弯的风险：(1)不对称指数高提示患者术前胸廓不对称，胸廓不对称可能导致脊柱受到不均衡的机械应力，从而引起脊柱侧弯[24]；(2)胸廓不对称会影响Nuss支架的放置和固定，如果支架放置不当或固定不牢，可能发生支架移位或脱落，进而引起脊柱侧弯；(3)胸廓不对称会影响术后胸廓的重塑过程，如果胸廓重塑不完全或不对称，可能对脊柱产生持续的异常应力，从而导致侧弯[9]。
本研究基于Cox分析结果初步建立了针对漏斗胸患者Nuss术后发生脊柱侧弯的风险预测模型，此模型可以帮助医生进行术前风险评估，对于高风险患者，医生可以更加慎重地考虑手术适应证和风险管理策略[25]。通过预测模型，患者可以获得个性化的术后脊柱侧弯风险评估结果[26]。但本研究也存在以下局限性：首先，为单中心回顾性研究，样本量相对较小，可能影响结果的可靠性和外推性，未来还需要开展多中心、大样本的前瞻性研究，以进一步验证本研究结果；其次，研究对象的平均随访时间短于预期，部分患者的随访时间不足，可能低估了Nuss术后脊柱侧弯的发生率，尽管敏感性分析显示随访时间的差异并未显著影响研究结论，但仍需延长随访时间以确认研究结果的长期稳定性；再次，研究仅纳入了Nuss术后脊柱侧弯的影像学指标，未评估患者的临床症状和生活质量等指标，将来的研究应综合考虑影像学和临床指标，以全面评估Nuss手术对脊柱侧弯的影响；最后，本研究建立的预测模型尚未经过外部验证，其泛化性和实际应用价值有待进一步评估，未来应在独立的人群中开展前瞻性研究，以验证该预测模型的外部适用性。
综上，Nuss手术对漏斗胸患者疗效显著，但术后脊柱侧弯并发症的发生比例较高。术前合并肺动脉高压及影像学指标中的不对称指数高是Nuss术后脊柱侧弯发生的独立影响因素，基于上述指标建立的模型能够有效预测患者术后发生脊柱侧弯的风险概率，对于指导临床个体化诊疗随访计划的制定和实施，具有重要的临床意义及价值。

利益冲突  所有作者均声明不存在利益冲突。

作者贡献声明  李博闻：实验设计，数据收集、分析和解释，论文撰写；张强：论文的审阅及把关，提供基金支持；孙益鑫：数据收集、分析和解释；所有作者均对最终文稿进行审读并确认。

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