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Abstract

Study Design

Original research, cross-sectional study.

Objectives

Evaluate preferences of patients, caregivers, and patient-caregiver dyads for non-fusion vs fusion surgery.

Methods

A survey-based discrete choice experiment included patients 12-15 years of age with idiopathic scoliosis and their caregivers. Preference weights were estimated for 2 types of attributes that differed between fusion and non-fusion surgery: treatment outcomes (ie, appearance, confidence in planned correction, spinal motion, device failure, reoperation, and recovery period) and procedure-type requirements associated with each surgery type. Correlations between preference weights for patients, caregivers, and dyads were calculated to assess preference concordance between cohorts.

Results

A total of 344 respondents (124 patients, 92 caregivers, and 128 patient/caregiver dyads) completed the survey. Appearance was the most important treatment outcome for the average respondent in all cohorts. For patients, the least important treatment outcome was recovery time, whereas reoperation rate was the least important treatment outcome for caregivers and dyads. After controlling for treatment outcomes, patients and patient-caregiver dyads preferred the procedure-type requirements associated with fusion surgery, while caregivers preferred the procedure-type requirements associated with non-fusion surgery. Preferences of respondents who completed the survey prior to surgery were similar to those who completed the survey after surgery. Patient and caregiver preferences were more similar to the preferences of dyads than to each other.

Conclusions

Patients and patient-caregiver dyads preferred the procedure-type requirements associated with fusion surgery, while caregivers preferred the procedure-type requirements associated with non-fusion surgery. Appearance was the most important treatment outcome for patients, caregivers, and patient-caregiver dyads.

Keywords

pediatric idiopathic scoliosis spinal deformity patient preference information discrete choice experiment non-fusion spinal surgery spinal fusion surgery vertebral body tether patient caregiver dyad

Introduction

Idiopathic scoliosis is defined as a lateral curvature of the spine ≥10° associated with vertebral rotation that lacks an identifiable cause and is the most common type of pediatric spinal deformity.¹ Idiopathic scoliosis is most frequently diagnosed between 10 to 18 years and is referred to as adolescent idiopathic scoliosis (AIS). Idiopathic scoliosis of 10° or more occurs in about 2.5% of most adolescent populations, however, in only about 0.25% does a curve progress to where treatment is warranted.^2,3 Nonoperative brace treatment is considered for skeletally immature patients with curves greater than 20-25°, while surgical treatment is considered for curves greater than 40-45° that are progressive despite brace treatment in skeletally immature patients, and for curves approaching or greater than 50° in skeletally mature patients.^4,5 The surgical treatment for AIS with the longest track record is instrumented posterior spinal fusion which provides immediate, excellent, and durable correction of spinal curvatures with low rates of complications including revision surgery.^6,7 However, fusion surgery results in permanent loss of motion and growth across the instrumented spinal levels and may lead to degeneration of adjacent spinal segments and reduced health-related quality of life in adulthood.^8-10 Non-fusion spinal devices including anterior vertebral body tethers (AVBT) and posterior dynamic distraction (PDD) have received marketing authorization by the United States Food and Drug Administration (FDA) for treatment of pediatric scoliosis.^11-13 These devices aim to correct spinal curvatures while preserving spinal growth, spinal mobility and delaying or preventing the need for spinal fusion.^14,15 While AVBT and PDD have been shown to delay or avoid spinal fusion in appropriately selected patients, complications and reoperations are higher compared to fusion, and spinal deformity correction has been reported as less consistent and predictable compared to fusion.^16-20 However, limited published data which compares long term outcomes of non-fusion vs fusion surgery for treatment of AIS is currently available to guide decision-making by patients and families.

Treatment of pediatric spinal deformity was recognized by the FDA as a preference-sensitive priority area for medical devices due to the patient benefit-risk tradeoffs and impact of uncertainty related to treatment decisions regarding non-fusion spinal surgery vs spinal fusion.²¹ In 2019 a multidisciplinary team comprised of health professionals including spine surgeons, researchers, a patient advocacy organization, and FDA staff developed a survey to identify the benefits, risks, and key attributes of fusion surgery and non-fusion surgery. This survey was designed as a discrete choice experiment (DCE) and collected quantitative patient preference information for a set of treatment related attributes associated with fusion surgery and non-fusion surgery (AVBT). Results of this initial study showed that a single cohort comprised of patients and their caregivers valued appearance and preservation of spinal motion and, to a lesser extent, reoperation rates when considering non-fusion vs fusion surgery.²² To our knowledge, this was the first study to quantitatively evaluate patient and parent preferences for fusion surgery compared to non-fusion surgery for treatment of adolescent idiopathic scoliosis using a discrete choice experimental design. This follow-up study was conducted to address the research question of whether differences in preferences exist for non-fusion vs fusion surgery from the perspectives of patients, caregivers, and patient-caregiver dyads. The aim of this study is to better understand how the preferences of pediatric patients, caregivers, and patient-caregiver dyads interact and influence decision-making, and to utilize these data to help inform future clinical and regulatory decision-making.

Materials and Methods

The preferences of pediatric patients, caregivers, and patient-caregiver dyads associated with fusion and non-fusion surgery were evaluated using an online DCE. DCEs elicit stated-preference information using a survey-based design that requires respondents to choose between profiles for a medical intervention that are defined in terms of attributes and various manifestations of the attributes (attribute levels) associated with a specific medical intervention. DCEs are based on the hedonic principle that choices between alternatives correspond with the relative importance of their features or attributes, and that systematic variations in choices can be leveraged as an indication of the relative importance (or preference) associated with attribute changes. The collection of these data is based on measurement theories in cognitive psychology.²³ The analysis of the choice data generated through DCEs is based on random-utility theory, which links choices and utility.²⁴

Survey Design

A multidisciplinary team developed a DCE in accordance with good research practices²⁵ and FDA guidance²⁶ using patient-centric plain language at an 8^th grade reading level. Survey attributes were identified based on a literature review, as well as input from the multidisciplinary team which included 8 spinal surgeons with extensive experience in pediatric scoliosis surgery. Table 1 shows the attributes and attribute levels used in the DCE choice tasks. Two broad attribute categories were identified. One category identified relevant treatment outcomes which included¹: confidence in the ability of the selected treatment to provide the planned correction of the spinal curve,² improvement in trunk (back) appearance 1 year after surgery (level of correction),³ motion within the treated spinal area,⁴ risk of the spinal device failing to work as expected,⁵ risk of another surgical procedure (reoperation), and⁶ the patient feeling “back to normal” (recovery period). The other attribute category was termed procedure-type requirements and was intended to capture all aspects of surgical treatment not represented by the 6 treatment outcome attributes. This attribute category referred to as procedure-type requirements represented the bundle of factors required by each of the 2 types of surgeries presented to respondents (ie, fusion surgery, and non-fusion surgery (AVBT)) and was captured under an alternative specific constant (ASC).

Table 1.

Survey Treatment Outcome Attributes and Attribute Levels for Fusion and Non-Fusion Scenarios Used to Generate Discrete-Choice Experiment Questions Presented to Respondents for Treatment Outcomes for Adolescent Idiopathic Scoliosis.

Survey Attribute	Fusion (Fixed Attribute Levels)	Non-fusion (Variable Attribute Levels)
Confidence of the selected treatment to provide the planned correction of the spinal curve	• 90% (excellent confidence)	• 60% (medium)
		• 80% (high)
		• 90% (excellent)
Improvement in trunk appearance (back appearance) 1 year after surgery	• Level 3 (excellent improvement)	• Level 1 (some improvement)
		• Level 2 (good improvement)
		• Level 3 (excellent improvement)
Motion within treated spinal area	• No motion	• All motion remains
		• Some motion remains
		• No motion
Risk of spinal device failing to work as expected	• 1%	• 1%
		• 20%
		• 40%
Risk of another surgery (reoperation)	• 3%	• 3%
		• 8%
		• 10%
Patient feeling back to normal (recovery)	• 6 weeks	• 2 weeks
		• 4 weeks
		• 6 weeks

Note: The discrete-choice experiment questions were labeled. This means every question presented a fusion and non-fusion option. While this label was not experimentally assigned to the alternatives, they represent an attribute meant to capture all aspects of each of the 2 types of surgeries presented to respondents (ie, fusion surgery, and non-fusion surgery (AVBT)) not represented by the outcomes in the table. We identify this attribute as procedure-type requirements.

As outcomes associated with fusion surgery for adolescent idiopathic scoliosis are well-established with a high level of certainty, a fixed profile was identified for the fusion attribute levels to reflect outcomes associated with posterior spinal fusion and posterior spinal instrumentation using contemporary surgical techniques: 90% confidence in planned correction of the spinal curve, excellent improvement in trunk (back) appearance (level 3), no motion after surgery within the treated spinal area, 1% risk of spinal device failure, 3% reoperation rate, and a 6-week recovery period. The non-fusion surgery attribute levels were varied and were defined to encompass the range of outcomes considered clinically plausible based on published literature and expert consensus among the 8 spinal surgeon members of the multidisciplinary team using a nominal group technique. The spinal surgeon investigators were orthopedic surgeons and neurosurgeons from a mix of academic and private practice settings widely distributed across the US and had extensive experience in both fusion and non-fusion surgery for pediatric scoliosis.

A draft survey was created and contained 3 parts. Part 1 collected basic demographic information (patient age, patient sex, caregiver educational level) and prior experiences of patients and caregivers with scoliosis. Part 2 provided a tutorial on the benefits and risks of surgery for idiopathic scoliosis and an overview of fusion surgery vs non-fusion surgery. Part 3 asked the respondents to choose between non-fusion and fusion surgery for various experimentally designed case profiles. The draft survey instrument was pretested in 8 interviews with patients and their caregivers that met the study inclusion criteria. The results from pretesting were used to refine the survey and ensure respondents comprehended the choice tasks and the information contained in the survey. Pretesting was also used to ensure that respondents were trading off between the outcomes.

The final DCE survey was a labeled design in which a fixed profile for fusion was compared to various experimentally designed case profiles in which the attribute levels for non-fusion varied (Figure 1). The design of the non-fusion surgery options relied on a D-efficient design with 420 questions divided into 60 blocks of 7 questions using Lighthouse Studio 9.1 (Sawtooth Software, Inc, Provo UT). Surveys were presented to the patient and caregiver together, and survey respondents identified who completed the survey (patient, caregiver, or both together).

Figure 1.

Example question from the survey-based discrete-choice experiment presented to respondents.

Survey Participants

The DCE was offered to all eligible patients and caregivers evaluated at 7 pediatric scoliosis spine centers distributed across the United States with expertise in both fusion and non-fusion surgery over a 1-year time period. Inclusion criteria were: (1) 10-21-year-old patients who were considering AIS surgery, (2) 10-21-year-old patients who had undergone either fusion surgery or non-fusion surgery for AIS in the past 3 years, and (3) caregivers of the AIS patients. Study participants were recruited in the clinic and the DCE was completed via an online platform either in the clinic space (for those without internet connectivity or online access) or at home via a website link without assistance from research or clinical personnel. Respondents were stratified into 3 cohorts based on who completed the survey: (1) DCE completed by the patient, (2) DCE completed by the caregiver, and (3) DCE completed jointly by the patient and caregiver (dyads). All research activities were Institutional Review Board (IRB) approved at the central study coordination site and at each participating site.

Data Analysis

Three random-parameters logit (RPL) models were used to analyze choices collected from each of 3 respondent groups. Each model regressed choices against differences in the profiles presented in each question. Preference weights were represented as log-odds indicating the rate at which the relative probability of choice changed when a profile included specific attribute levels, all else equal.²⁷ Cohort-specific preference weights were estimated.

Attribute relative-importance weights for each cohort were calculated from the preference weights for each outcome in the DCE. Preference weights are relative in nature and are not directly interpretable. However, greater differences between preference weights indicate greater changes in the probability of choice. Relative importance weights indicate the maximum impact that changes in the study attributes had on the likelihood of choosing a treatment option in the DCE. The sum of the overall importance of attributes was normalized to add up to 1 within groups.²⁸

The impact of previous patient experience with fusion or non-fusion surgery was evaluated by including whether this experience affected the relative importance of procedure-type requirements. This was accomplished by generating an interaction term between a dummy-coded variable indicating whether the patient had already completed surgery for scoliosis or not, and the ASC capturing the type of surgery respondents could choose (ie, fusion or non-fusion). New cohort-specific preference models were estimated including the interaction term between the ASC and the patient timing for survey completion (ie, before or after surgery). Significance of this interaction would indicate that completion of the survey after surgery was associated with systematically different preferences for the procedure-type requirements. To assess the magnitude of the interaction effect, we used the estimates with the interaction term to generate expected choice probabilities (ECPs) between fusion and non-fusion surgery by cohort and surgery experience. These ECPs represent the probability of choosing 1 surgery over another by the average respondent in the cohort and experience group, assuming all outcomes are expected to be the same. This had the effect of isolating the impact of procedure-type requirement on choice. Lastly, pairwise correlations of preference weights across cohorts were calculated using a simple Pearson’s correlation coefficient.

Results

Demographics

A total of 345 respondents started and 344 respondents fully completed the DCE and therefore 344 respondents were utilized in the analysis. Of the total of 344 respondents, 36.1% (n = 124) were patients, 26.7% (n = 92) were caregivers and 37.2% (n = 128) were patient/caregiver dyads. Mean patient age was 15.2 years (SD 3.1 years) and 82.5% were female. One hundred seventy-three respondents were enrolled prior to surgery, and 171 were enrolled after surgery (99 after fusion, 71 after non-fusion surgery, 1 not-reported, Table 2).

Table 2.

Survey Respondent Demographics for Adolescent Idiopathic Scoliosis Discrete Choice Experiment Survey.

Characteristic	Pre-operative	Post-operative
	N = 173	N = 171	N = 344
	Count (%)*	Count (%)*	Count (%)*
Respondent type
Patients	52 (30.1)	72 (42.1)	124 (36.1)
Caregivers	52 (30.1)	40 (23.4)	92 (26.7)
Caregivers and patients completed survey together	69 (39.8)	59 (34.5)	128 (37.2)
Patient age, mean (SD)	15.7 (3.2)	16.7 (3.0)	16.2 (3.1)
Educational attainment
None completed	46 (26.6)	48 (28.1)	94 (27.3)
High school graduate or equivalent	22 (12.7)	33 (19.3)	55 (16.0)
Trade/technical/vocational training	4 (2.3)	10 (5.9)	14 (4.1)
Associate degree	14 (8.1)	17 (9.9)	31 (9.0)
Bachelor’s degree	43 (24.9)	33 (19.3)	76 (22.1)
Graduate or professional degree	44 (25.4)	30 (17.5)	74 (21.5)
Previous surgery type (among post-operative respondents)			n = 171
Fusion		99 (57.8)	99 (57.8)
Non-fusion		71 (41.5)	71 (41.5)
Not reported		1 (0.5)	1 (0.5)
Patient sex
Female	146 (84.4)	137 (80.1)	283 (82.3)
Male	26 (15.0)	32 (18.7)	58 (16.9)
Other	0 (0)	1 (0.6)	1 (0.3)
Prefer not to answer	1 (0.6)	1 (0.6)	2 (0.6)

Relative Preference for Procedure Type Across Respondent Cohorts

Relative preference for procedure type (Table 3) was assessed by calculating the probability that the average respondent in each cohort would have chosen fusion vs non-fusion surgery assuming the outcomes from the 2 procedures were the same. On average, patients and patient-caregiver dyads preferred fusion surgery, although for dyads this preference was not statistically significant at a 95% confidence level, while caregivers preferred non-fusion surgery.

Table 3.

Procedure-Type Requirements: Predicted Choice Probabilities by Cohort for Fusion and Non-Fusion Surgery.

	Patients (n = 124)	Caregivers (n = 92)	Dyads (n = 128)
Fusion	94.38%	23.50%	77.86%
Fusion	(87.52% - 101.24%)^a	(−25.69% to 72.68%)^a	(24.43% - 131.3%)^a
Nonfusion	5.62%	76.50%	22.14%
Nonfusion	(−1.24% to 12.48%)^a	(27.32% - 125.69%)^a	(−31.3% to 75.57%)^a

^a95% confidence intervals are in parentheses.

Treatment Outcome Relative Importance Weights for Different Respondent Cohorts

Attribute relative importance weights for each cohort were calculated for treatment outcomes excluding procedure-type requirements (Table 4). The average respondent in all cohorts found appearance to be the most important treatment outcome. In the case of patients and dyads, this was followed in importance by device failure rate, whereas for caregivers the second most important attribute was confidence in planned correction. Patients were least concerned about recovery time, whereas caregivers and dyads were least concerned about reoperation rates.

Table 4.

Treatment Outcome Relative Importance Weights for Patient, Caregiver, and Patient-Caregiver Dyad Cohorts (Excludes Procedure-Type Requirements).

Patients	Mean Importance Weight (SE)	Caregivers	Mean Importance Weight (SE)	Dyads	Mean Importance Weight (SE)
Appearance	0.19 (0.05)	Appearance	0.31 (0.09)	Appearance	0.36 (0.09)
Device failure	0.16 (0.06)	Confidence in planned correction	0.21 (0.08)	Device failure	0.22 (0.08)
Reoperation	0.15 (0.05)	Device failure	0.14 (0.06)	Recovery	0.1 (0.06)
Motion	0.12 (0.03)	Recovery	0.13 (0.06)	Motion	0.1 (0.01)
Confidence in planned correction	0.03 (0.06)	Motion	0.11 (0.01)	Confidence in planned correction	0.09 (0.06)
Recovery	0.01 (0.06)	Reoperation	0.01 (0.05)	Reoperation	0.08 (0.04)

Correlations of Preference Weights for Procedure-Type Requirements Across Cohorts

All correlation values are positive suggesting that overall preference estimates for these cohorts move in the same direction (Table 5). Patient preference estimates for procedure type requirements are more highly correlated with the preference estimates of dyads (Rho = 0.69) than with the preferences of caregivers (Rho = 0.39). Caregiver preferences are also more highly correlated with the preferences of dyads (Rho = 0.90) than with the preferences of patients (Rho = 0.39).

Table 5.

Correlations of Preference Weights for Procedure-type Requirements for Patient, Caregiver, and Patient-Caregiver Dyad Cohorts.

	Patients	Caregivers	Dyads
Patients	1.00
Caregivers	0.39	1.00
Dyads	0.69	0.90	1.00

Impact of Completing the Survey Before or After Surgery

The impact of taking the survey after surgery was evaluated in terms of changes in respondents’ ECPs for fusion or non-fusion procedure-type requirements (Table 6). Patient and caregiver preferences for procedure type were relatively stable between those who completed the survey before or after surgery as the ECPs for each group remained largely constant among those who completed the survey before and after the procedure. The average patients with and without previous scoliosis surgery were expected to choose fusion surgery by a wide margin based solely on the procedure-type requirements. Among caregivers, the ECPs are also stable before and after surgery, but the average member of this group is predicted to prefer non-fusion procedure-type requirements. For respondents who answered the DCE in dyads, completing the survey before surgery was associated with an expected choice distribution that mirrors the expected patients’ choices. In contrast, for dyads who completed the survey after surgery the ECPs between the 2 procedures are more evenly distributed.

Table 6.

Predicted Choice Probability by Cohort and Previous Experience With Scoliosis Surgery Based on Procedure-Type Requirements.

	Survey Completed Before Surgery
	Patient (n = 52)	Caregivers (n = 52)	Dyads (n = 69)
Fusion	91.49%	16.91%	99.95%
Fusion	(73.71%-109.27%)^a	(-34.05% to 67.86%)^a	(99.69%-100.21%)^a
Nonfusion	8.51%	83.09%	0.05%
Nonfusion	(−9.27% to 26.29%)^a	(32.14%-134.05%)^a	(−0.214% to 0.312%)^a

	Survey completed after surgery
	Patient (n = 72)	Caregivers (n = 40)	Dyads (n = 59)
Fusion	95.15%	3.43%	54.22%
Fusion	(84.09%-106.21%)^a	(−7.56% to 14.41%)^a	(−20.92% to 129.36%)^a
Nonfusion	4.85%	96.57%	45.78%
Nonfusion	(−6.21% to 15.91%)^a	(85.59%-107.56%)^a	(−29.36% to 120.92%)^a

^a95% confidence interval.

Discussion

In our study, notable differences were observed among the 3 study cohorts regarding relative preference for fusion vs non-fusion surgery and the attributes that drove these choices. On average, patients and patient-caregiver dyads preferred the procedure-type requirements associated with fusion surgery, while caregivers preferred non-fusion surgery. The average respondent in each cohort identified appearance as the most important treatment outcome attribute that impacted the likelihood of choosing a fusion or non-fusion surgery. For patients and patient-caregiver dyads, the second most important treatment outcome attribute was device failure rate, whereas for caregivers the second most important treatment outcome attribute was confidence in planned correction. For patients, relative preference weights were least influenced by changes in recovery time, whereas relative preference weights of caregivers and patient-caregiver dyads were least influenced by changes in reoperation rates. Correlations of preference weights across the 3 cohorts for procedure-type requirements demonstrated that patient preferences were more highly correlated with the preference of dyads than with the preferences of caregivers. Caregiver preferences are also more highly correlated with the preferences of dyads than with the preferences of patients. These correlations between attribute preference weights of both caregivers and patients with the preference weights of dyads support the result that caregivers are more likely driving the discussions about surgery type when the decisions are considered in dyads. Predicted choices by surgery type for patients and caregivers appeared stable when completing the survey before or after surgery. For dyads, results show that respondents who completed the survey prior to surgery strongly preferred fusion when considering only the procedure-type requirements, while the ECPs for respondents who completed the survey after surgery were more equally apportioned between fusion and non-fusion.

Prior literature regarding preferences related to surgery for adolescent idiopathic scoliosis has focused on key attributes identified for posterior fusion surgery from the perspectives of patients, parents, and health care providers²⁹ and evaluation of parents’ and patients’ preferences and concerns regarding anterior, posterior, and combined fusion surgery.³⁰ Our study is the first to quantitatively explore preferences for treatment of adolescent idiopathic scoliosis with fusion vs non-fusion surgery from the perspectives of patients, caregivers, and patient-caregiver dyads using the same survey instrument. The present study also offers a novel approach to assess the decision dynamics between patients and caregivers and sheds light on the decision makers who are influencing treatment decisions in this population. Medical decision-making regarding AIS surgery is complex due to proxy decision-making by caregivers and the developmental maturation and increasing decision-making capabilities of adolescent patients over time which allows for their inclusion in treatment decisions.³¹ Surgery for AIS is most frequently undertaken to address concerns regarding body image due to trunk asymmetry and to prevent future adverse consequences related to curve progression including back pain, increased deformity, and pulmonary symptoms in patients with large thoracic curves. However, long term natural history studies have shown that the majority of patients with untreated moderate AIS function well in society as adults with respect to employment, marriage including childbearing, and maintenance of activity levels into older adulthood.³² Both fusion and non-fusion treatment of AIS are not without risks which include postoperative pain, infection, device failure, reoperation, and neurologic injury. Balancing the acute and long-term benefits and risks of surgical intervention pose challenges for adolescents and their caregivers when treatment preferences and perspectives on the roles each has in decision making differ,³³ particularly in light of concerns about respecting adolescents’ autonomy.³⁴ Priorities of each stakeholder might differ, with adolescents potentially more concerned about short-term outcomes, whereas caregivers might focus on long-term impacts of treatment decisions.³⁵ There exists a need for additional future research regarding intergenerational decision making related to treatments for AIS³⁶ and the authors are currently developing a shared decision-making tool as a pragmatic way to facilitate communication among patients, caregivers, and clinicians considering surgical treatment.

This study has several notable strengths and limitations. Regarding strengths, this study was designed by a multidisciplinary team comprised of a patient advocacy group, scoliosis experts, patients with scoliosis and their families, and FDA staff, and undertaken in accordance with recommended best practices for patient-preference studies. The study included both preoperative and postoperative patients with AIS who were considering surgical options as well as patients who had undergone fusion or non-fusion surgery in order to evaluate the diverse range of patients encountered in clinical practice. Limitations of this study included variability in administration of the DCE as it was completed by either the patient, caregiver or patient-caregiver dyad, and separate surveys were not completed by each, and versions of the survey were not randomized across respondents. This cross-sectional study design was selected as it was considered representative of real-world decision-making and minimized respondent burden and disruption of clinical workflow. Also, the patients and caregivers who entered this study were selected from scoliosis centers where surgeons had extensive experience performing both fusion and non-fusion surgery which may not be representative of all hospitals where scoliosis surgery is performed. Lastly, decisions regarding surgery were subject to influence by a range of sources including discussions with clinicians involved in the study and their care team members, non-study health care providers, other patients and family members, the internet and social media. It is not possible to determine how informed respondents were about the treatment options when they completed the survey, but in general, patients and their caregivers had access to many resources including scoliosis patient education web sites and web-based patient support groups. We attempted to minimize disparities or gaps in respondents’ understanding of their options by providing pre-survey education.

Conclusions

Patients and patient-caregiver dyads preferred the procedure-type requirements associated with fusion surgery, while caregivers preferred the procedure-type requirements associated with non-fusion surgery. The most important treatment outcome for patients, caregivers, and patient-caregiver dyads related to surgery for adolescent idiopathic scoliosis was trunk (back) appearance after surgery.

Footnotes

Acknowledgments

The Harms Study Group Investigators include Aaron Buckland, MD, Melbourne Orthopaedic Group & Royal Childrens Hospital; Ahmet Alanay, MD, Acibadem Maslak Hospital; Amer Samdani, MD, Shriners Hospitals for Children, Philadelphia; Amit Jain, MD, Johns Hopkins Hospital; Baron Lonner, MD, Mount Sinai Hospital; Benjamin Roye, MD, Columbia University; Bob Cho, MD, Shriners Hospitals for Children, Pasadena CA; Burt Yaszay, MD, Seattle Children’s Hospital; Caglar Yilgor, MD, Acibadem Maslak Hospital; Dan Hoernschmeyer, MD, University of Missouri Health Care; Daniel Hedequist, MD, Boston Children’s Hospital; Daniel Sucato, MD, Texas Scottish Rite Hospital; David Clements, MD, Cooper Bone & Joint Institute; Firoz Miyanji, MD, BC Children’s Hospital; Harry Shufflebarger, MD, Paley Orthopedic & Spine Institute; Jack Flynn, MD, Children’s Hospital of Philadelphia; Jean Marc Mac-Thiong, MD, CHU Sainte-Justine; Josh Murphy, MD, Children’s Healthcare of Atlanta; Joshua Pahys, MD, Shriners Hospitals for Children Philadelphia; Keith Bachmann, MD, University of Virginia; Kevin Neal, MD, Nemours Children’s Clinic Jacksonville; Laurel Blakemore, MD, Pediatric Specialists of Virginia/Children’s National; Lawrence Haber, MD, Ochsner Health Center for Children New Orleans; Lawrence Lenke, MD, Columbia University; Mark Abel, MD, University of Virginia; Mark Erickson, MD, Children’s Hospital Colorado; Michael Glotzbecker, MD, Rainbow Children’s Hospital; Michael Kelly, MD, Rady Children’s Hospital; Michael Vitale, MD, Columbia University; Michelle Marks, PT, MA, Setting Scoliosis Straight Foundation; Munish Gupta, MD, Washington University; Nicholas Fletcher, MD, Emory University; A. Noelle Larson, MD, Mayo Clinic; Patrick Cahill, MD, Children’s Hospital of Philadelphia; Paul Sponseller, MD, Johns Hopkins Hospital; Peter Gabos, MD, Nemours/Alfred I. duPont Hospital for Children; Peter Newton, MD, Rady Children’s Hospital; Peter Sturm, MD, Cincinnati Children’sHospital; Randal Betz, MD, Institute for Spine & Scoliosis; Stefan Parent, MD, CHU Sainte-Justine; Stephen George, MD, Nicklaus Children’s Hospital; Steven Hwang, MD, Shriners Hospitals for Children Philadelphia; Suken Shah, MD, Nemours/Alfred I. duPont Hospital for Children; Sumeet Garg, MD, Children’s Hospital Colorado; Tom Errico, MD, Nicklaus Children’s Hospital; and Vidyadhar Upasani, MD, Rady Children’s Hospital.

Declaration of conflicting interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

Funding was provided through FDA BAA- HDDISWP#83. NOTE: In addition to the above funding, this study was funded in part by grants to the Setting Scoliosis Straight Foundation in support of Harms Study Group research from DePuy Synthes Spine, EOS imaging/Atec, Stryker Spine, Medtronic, NuVasive, ZimVie, and Biedermann Motech.

IRB approval

IRB approval was obtained for all aspects of this study (Western IRB study number 1281165, tracking number 20200565).

Ethical Statement

ORCID iDs

Vincent J. Devlin

A. Noelle Larson

Peter Newton

Baron Lonner

Data Availability Statement

Authors are not authorized for participation in research data sharing.*

References

Asher

Burton

. Adolescent idiopathic scoliosis: natural history and long term treatment effects. Scoliosis. 2006;1(1):2. doi:10.1186/1748-7161-1-2

Weinstein

. Natural history. Spine. 1999;24(24):2592-2600. doi:10.1097/00007632-199912150-00006

US Preventive Services Task Force Grossman

Curry

, et al. Screening for adolescent idiopathic scoliosis: US Preventive Services Task Force recommendation statement. JAMA. 2018;319(2):165-172. doi:10.1001/jama.2017.19342

Weinstein

Dolan

Wright

Dobbs

. Effects of bracing in adolescents with idiopathic scoliosis. N Engl J Med. 2013;369(16):1512-1521. doi:10.1056/NEJMoa1307337

Lenke

Kuklo

Ondra

Polly

Jr . Rationale behind the current state-of-the-art treatment of scoliosis (in the pedicle screw era). Spine. 2008;33(10):1051-1054. doi:10.1097/BRS.0b013e31816f2865

Ahmed

Bastrom

Yaszay

Newton

Harms Study Group . 5-year reoperation risk and causes for revision after idiopathic scoliosis surgery. Spine. 2017;42(13):999-1005. doi:10.1097/BRS.0000000000001968

Newton

Ohashi

Bastrom

, et al. Prospective 10-year follow-up assessment of spinal fusions for thoracic AIS: radiographic and clinical outcomes. Spine Deform. 2020;8(1):57-66. doi:10.1007/s43390-019-00015-1

Hariharan

Shah

Petfield

, et al. Complications following surgical treatment of adolescent idiopathic scoliosis: a 10-year prospective follow-up study. Spine Deform. 2022;10(5):1097-1105. doi:10.1007/s43390-022-00508-6

Marks

Newton

Petcharaporn

, et al. Postoperative segmental motion of the unfused spine distal to the fusion in 100 patients with adolescent idiopathic scoliosis. Spine. 2012;37(10):826-832. doi:10.1097/BRS.0b013e31823b4eab

10.

Lonner

Ren

Upasani

, et al. Disc degeneration in unfused caudal motion segments ten years following surgery for adolescent idiopathic scoliosis. Spine Deform. 2018;6(6):684-690. doi:10.1016/j.jspd.2018.03.013

11.

U.S. Food and Drug Administration . The Tether™ - vertebral body tethering system (H190005). 2019. https://www.accessdata.fda.gov/scripts/cdrh/cfdocs/cfhde/hde.cfm?id=H190005

12.

U.S. Food and Drug Administration . Minimally invasive deformity correction (MID-C) system (H170001). 2019. https://www.accessdata.fda.gov/scripts/cdrh/cfdocs/cfhde/hde.cfm?id=403563

13.

U.S. Food and Drug Administration . REFLECT™ scoliosis correction system (H210002). 2023. https://www.accessdata.fda.gov/scripts/cdrh/cfdocs/cfHDE/hde.cfm?id=490516

14.

Pahys

Samdani

Hwang

Warshauer

Gaughan

Chafetz

. Trunk range of motion and patient outcomes after anterior vertebral body tethering versus posterior spinal fusion: comparison using computerized 3D motion capture technology. J Bone Joint Surg Am. 2022;104(17):1563-1572. doi:10.2106/JBJS.21.00992

15.

McDonald

Shah

Hargiss

, et al. When successful, anterior vertebral body tethering (VBT) induces differential segmental growth of vertebrae: an in vivo study of 51 patients and 764 vertebrae. Spine Deform. 2022;10(4):791-797. doi:10.1007/s43390-022-00471-2

16.

Shaw

Welborn

Matsumoto

, et al. To tether or fuse? Significant equipoise remains in treatment recommendations for idiopathic scoliosis. Spine Deform. 2022;10(4):763-773. doi:10.1007/s43390-022-00497-6

17.

Shin

Arguelles

Cahill

Flynn

Baldwin

Anari

. Complications, reoperations, and mid-term outcomes following anterior vertebral body tethering versus posterior spinal fusion: a Meta-Analysis. JB JS Open Access. 2021;6(2):e2100002. doi:10.2106/JBJS.OA.21.00002

18.

Newton

Parent

Miyanji

, et al. Anterior vertebral body tethering compared with posterior spinal fusion for major thoracic curves: a retrospective comparison by the Harms Study Group. J Bone Joint Surg Am. 2022;104(24):2170-2177. doi:10.2106/JBJS.22.00127

19.

Raitio

Syvänen

Helenius

. Vertebral body tethering: indications, surgical technique, and a systematic review of published results. J Clin Med. 2022;11(9):2576. doi:10.3390/jcm11092576

20.

Todderud

Milbrandt

Floyd

, et al. Preliminary study of motion preservation following posterior dynamic distraction device in adolescent idiopathic scoliosis patients. J Pediatr Orthop. 2024;44(9):524-529. doi:10.1097/BPO.0000000000002739

21.

U.S. Food and Drug Administration . List of patient preference-sensitive priority areas. https://www.fda.gov/about-fda/cdrh-patient-science-and-engagement-program/list-patient-preference-sensitive-priority-areas#tradeoffs

22.

Larson

Marks

Gonzalez Sepulveda

, et al.

Non-fusion versus fusion surgery in pediatric idiopathic scoliosis: what trade-offs in outcomes are acceptable for the patient and family?

J Bone Joint Surg Am. 2024;106(1):2-9. doi:10.2106/JBJS.23.00503

23.

Thurstone

. A law of comparative judgment. Psychol Rev. 1927;34(4):273-286. doi:10.1037/h0070288

24.

McFadden

. Conditional logit analysis of qualitative choice behavior. UC Berkeley: Institute of Urban and Regional Development, 1-48; 1972. https://escholarship.org/uc/item/61s3q2xr

25.

Bridges

JFP

de Bekker-Grob

Hauber

, et al. A roadmap for increasing the usefulness and impact of patient-preference studies in decision making in health: a good practices report of an ISPOR task force. Value Health. 2023;26(2):153-162. doi:10.1016/j.jval.2022.12.004

26.

U.S. Food and Drug Administration . Patient preference information - Voluntary submission, review in premarket approval applications, humanitarian device exemption applications, and de novo requests, and inclusion in decision summaries and device labeling - guidance for industry. Food and Drug Administration staff, and other stakeholders. 2016. https://www.fda.gov/media/92593/download

27.

Hauber

González

Groothuis-Oudshoorn

, et al. Statistical methods for the analysis of discrete choice experiments: a report of the ISPOR conjoint analysis good research practices task force. Value Health. 2016;19(4):300-315. doi:10.1016/j.jval.2016.04.004

28.

Gonzalez

. A guide to measuring and interpreting attribute importance. Patient. 2019;12(3):287-295. doi:10.1007/s40271-019-00360-3

29.

Bunch

Chapman

. Patient preferences in surgery for scoliosis. J Bone Joint Surg Am. 1985;67(5):794-799.

30.

Bridwell

Shufflebarger

Lenke

Lowe

Betz

Bassett

. Parents' and patients' preferences and concerns in idiopathic adolescent scoliosis: a cross-sectional preoperative analysis. Spine. 2000;25(18):2392-2399. doi:10.1097/00007632-200009150-00020

31.

Katz

Webb

COMMITTEE ON BIOETHICS . Informed consent in decision-making in pediatric practice. Pediatrics. 2016;138(2):e20161485. doi:10.1542/peds.2016-1485

32.

Weinstein

. The natural history of adolescent idiopathic scoliosis. J Pediatr Orthop. 2019;39(Issue 6):S44-S46. doi:10.1097/BPO.0000000000001350

33.

Lin

Lipstein

Wittenberg

, et al. Intergenerational decision making: the role of family relationships in medical decision making. MDM Policy Pract. 2021;6(2):23814683211039468. doi:10.1177/23814683211039468

34.

Marron

. Adolescent shared decision-making: where we have been and where we are going. J Adolesc Health. 2021;69(1):6-7. doi:10.1016/j.jadohealth.2021.04.009

35.

Lipstein

Brinkman

Britto

. What is known about parents' treatment decisions? A narrative review of pediatric decision making. Med Decis Mak. 2012;32(2):246-258. doi:10.1177/0272989X11421528

36.

Ifelayo

Brito

Hargraves

Larson

. Development of a shared decision-making tool for adolescents with scoliosis to decide between observation versus fusion surgery. J Pediatr Orthop. 2021;41(Suppl 1):S70-S74. doi:10.1097/BPO.0000000000001800

Fusion versus Non-fusion Surgery for Pediatric Idiopathic Scoliosis: Preferences of Patients,Caregivers,and Patient-Caregiver Dyads

Abstract

Study Design

Objectives

Methods

Results

Conclusions

Keywords

Introduction

Materials and Methods

Survey Design

Survey Participants

Data Analysis

Results

Demographics

Relative Preference for Procedure Type Across Respondent Cohorts

Treatment Outcome Relative Importance Weights for Different Respondent Cohorts

Correlations of Preference Weights for Procedure-Type Requirements Across Cohorts

Impact of Completing the Survey Before or After Surgery

Discussion

Conclusions

Footnotes

Acknowledgments

Declaration of conflicting interests

Funding

IRB approval

Ethical Statement

ORCID iDs

Data Availability Statement

References