Digitala Vetenskapliga Arkivet

Change search
CiteExportLink to record
Permanent link

Direct link
Cite
Citation style
  • apa
  • ieee
  • modern-language-association-8th-edition
  • vancouver
  • Other style
More styles
Language
  • de-DE
  • en-GB
  • en-US
  • fi-FI
  • nn-NO
  • nn-NB
  • sv-SE
  • Other locale
More languages
Output format
  • html
  • text
  • asciidoc
  • rtf
Prognostic model development for risk of curve progression in adolescent idiopathic scoliosis: a prospective cohort study of 127 patients
Linköping University, Faculty of Medicine and Health Sciences. Linköping University, Department of Health, Medicine and Caring Sciences, Division of Prevention, Rehabilitation and Community Medicine.ORCID iD: 0000-0002-3639-6380
Department of Clinical Science, Intervention and Technology (CLINTEC), Division of Orthopaedics and Biotechnology, Karolinska Institutet, Stockholm; Department of Reconstructive Orthopaedics, Karolinska University Hospital Huddinge, Stockholm, Sweden .ORCID iD: 0000-0001-5228-738X
Department of Clinical Science, Intervention and Technology (CLINTEC), Division of Orthopaedics and Biotechnology, Karolinska Institutet, Stockholm; Department of Reconstructive Orthopaedics, Karolinska University Hospital Huddinge, Stockholm, Sweden .
Linköping University, Faculty of Medicine and Health Sciences. Linköping University, Department of Health, Medicine and Caring Sciences, Division of Prevention, Rehabilitation and Community Medicine.ORCID iD: 0000-0001-8612-583X
Show others and affiliations
2024 (English)In: Acta Orthopaedica, ISSN 1745-3674, E-ISSN 1745-3682, Vol. 95, p. 536-544Article in journal (Refereed) Published
Abstract [en]

Background and purpose: The study’s purpose was to develop and internally validate a prognostic survival model exploring baseline variables for adolescent idiopathic scoliosis curve progression.

Methods: A longitudinal prognostic cohort analysis was performed on trial data (n = 135) including girls and boys, Cobb angle 25–40°, aged 9–17 years, remaining growth > 1 year, and previously untreated. Prognostic outcome was defined as curve progression of Cobb angle of > 6° prior to skeletal maturity. 34 candidate prognostic variables were tested. Time-to-event was measured with 6-month intervals. Cox proportional hazards regression survival model (CoxPH) was used for model development and validation in comparison with machine learning models (66.6/33.3 train/test data set). The models were adjusted for treatment exposure.

Results: The final primary prognostic model included 127 patients, predicting progress with acceptable discriminative ability (concordance = 0.79, 95% confidence interval [CI] 0.72–0.86). Significant prognostic risk factors were Risser stage of 0 (HR 4.6, CI 2.1–10.1, P < 0.001), larger major curve Cobb angle (HRstandardized 1.5, CI 1.1–2.0, P = 0.005), and higher score on patient-reported pictorial Spinal Appearance Questionnaire (pSAQ) (HRstandardized 1.4, CI 1.0–1.9, P = 0.04). Treatment exposure, entered as a covariate adjustment, contributed significantly to the final model (HR 3.1, CI 1.5–6.0, P = 0.001). Sensitivity analysis displayed that CoxPH maintained acceptable discriminative ability (AUC 0.79, CI 0.65–0.93) in comparison with machine learning algorithms.

Conclusion: The prognostic model (Risser stage, Cobb angle, pSAQ, and menarche) predicted curve progression of > 6° Cobb angle with acceptable discriminative ability. Adding patient report of the pSAQ may be of clinical importance for the prognosis of curve progression.

Place, publisher, year, edition, pages
MJS Publishing, Medical Journals Sweden AB , 2024. Vol. 95, p. 536-544
Keywords [en]
Adolescent Idiopathic Scoliosis, Curve progression, Discriminative ability, Internal validation, Prognosis
National Category
Cancer and Oncology
Identifiers
URN: urn:nbn:se:liu:diva-207679DOI: 10.2340/17453674.2024.41911ISI: 001314905300004PubMedID: 39287215OAI: oai:DiVA.org:liu-207679DiVA, id: diva2:1898205
Available from: 2024-09-17 Created: 2024-09-17 Last updated: 2024-10-07
In thesis
1. Adolescent Idiopathic Scoliosis: Postural Stability, Prognostic factors and Impact of Conservative Treatments on Radiologic, Clinical and Self-Reported Outcomes
Open this publication in new window or tab >>Adolescent Idiopathic Scoliosis: Postural Stability, Prognostic factors and Impact of Conservative Treatments on Radiologic, Clinical and Self-Reported Outcomes
2024 (English)Doctoral thesis, comprehensive summary (Other academic)
Abstract [en]

Background: Adolescent idiopathic scoliosis (AIS) is a three-dimensional distortion of spinal curvature of unknown cause that develops during puberty. If the frontal plane curvature exceeds a Cobb angle of 24°, full-time bracing is recommended to prevent curve progression, negative health consequences or need for surgery. However, many patients decline full-time bracing, and limited evidence exists regarding alternative conservative treatments. Increasing knowledge of risk factors enables better identification of high-risk patients, thereby reducing the risk of over- or undertreatment.  

The overall aim of this thesis was to evaluate evidence of association between scoliosis and postural stability to inform a potential treatment target, to investigate the effectiveness of three alternative conservative treatments for patients who declined full-time bracing, and to develop a prognostic model for future risk of increased curvature in AIS.  

Methods: Data collection is based on one systematic literature review and meta-analysis, and one randomised controlled trial (RCT) for patients with AIS. In Paper I, literature was reviewed, and utilized posturography data to assess static postural stability to identify potential differences between patients with AIS and the control group (CON). In Papers II and III, patients were randomly assigned to either physical activity combined with hypercorrective Boston scoliosis night brace (NB), scoliosis-specific exercises (SSE), or a control group with physical activity alone (PA). Effects on trunk rotation, Cobb angle, self-reported physical activity, spinal appearance, and health-related quality of life were evaluated. Likewise, adherence, capability, and motivation in performing self-managed treatment were assessed. Follow-up was conducted at 6 months intervals until endpoint. The endpoint was defined as non-progression if the curvature increased by 6° or less at skeletal maturity, i.e., less than 1cm of growth over 6 months, or progression if the curvature increased by more than 6° before skeletal maturity. In Paper IV, data from the RCT were used for a longitudinal cohort analysis, developing and validating a prognostic model using Cox Proportional Hazards survival analysis. 

Results: Paper I found reduced postural stability in AIS patients compared to CON with increased sway area, mediolateral and anteroposterior range, and a posterior shift in the sagittal plane. The RCT included 135 individuals, mean age of 12.7 years (SD1.4) and Cobb angle of 31° (SD5.3). At 6 months, patients reported high adherence (72-95%) and motivation (65-92%) to the treatment, particularly in the NB group. Both the SSE and PA groups increased physical activity levels compared to the NB group. At endpoint, adherence remained adequate, but better in the NB and PA groups compared with the SSE group (50-89%). Adherence increased with higher capability and motivation, which explained 53% of the variance in adherence at endpoint. The SSE group had higher proportion of moderate problems in mobility and usual activities than the NB and PA groups, with no other clinically relevant between-group differences. The prognostic model (n=127) for curve progression showed acceptable discriminative ability (0.791), with risk factors including skeletal immaturity, larger major curve, and worsening spinal appearance. The model was adjusted for treatment exposure, as NB reduced the risk of curve progression.  

Conclusions: Patients with AIS have decreased postural stability with a posterior positional shift. Treatment evaluation at endpoint showed few between-group differences. However, the SSE group had higher proportion of moderate problems in mobility and usual activities compared to the other groups. Treatment adherence was adequate, but better in the NB and PA groups. Higher capability and motivation increased long-term adherence to the treatment. The treatment alternatives could be considered as the first option following a clinical decision on treatment. Including self-reported spinal appearance as a risk factor in the prognostic model may be clinically important for predicting which patients are at risk of curve progression. 

Abstract [sv]

Bakgrund: Idiopatisk skolios (AIS) är en tredimensionell ryggradskrökning som uppstår under puberteten utan känd orsak. Om kröken i frontalplanet når en Cobbsvinkel över 24 grader rekommenderas korsettbehandling under större delen av dygnet för att förhindra att kröken blir större och leder till negativa hälsokonsekvenser eller kirurgi. Många har svårt att acceptera dygnskorsett och det saknas kunskap om alternativa icke-kirurgiska behandlingsmetoder. Ökad kunskap om riskfaktorer möjliggör bättre identifiering av högriskpatienter och minskar därmed risken för över- eller underbehandling.  

Övergripande syfte med avhandlingen var att utvärdera evidens om sambandet mellan skolios och postural stabilitet vilket skulle kunna påverka behandlingen, att undersöka effektiviteten avseende tre alternativa icke kirurgiska behandlingar för patienter som avböjt dygnskorsett, samt att utveckla en prognostisk modell för framtida risk av ökad krök vid AIS.  

Metod: Datainsamlingen baserades på en systematisk litteraturöversikt med metaanalys och en randomiserad kontrollerad studie (RCT) för patienter med AIS. Delarbete I granskade litteratur och sammanvägde data från posturografi där postural stabilitet undersöktes för att kunna diagnostisera möjliga skillnader mellan patienter med AIS jämfört med kontrollgrupp (CON). I delarbete II och III lottades patienterna till endera fysisk aktivitet i kombination med nattkorsett (NB), eller skolios-specifik träning (SSE), eller kontrollgrupp med enbart fysisk aktivitet (PA). Effekter på bålrotation, Cobbsvinkel, samt självrapporterad fysisk aktivitet, ryggens utseende och hälso-relaterad livskvalité utvärderades. Likaså följsamhet, motivation och egenförmåga att genomföra behandlingen. Uppföljning utfördes var sjätte månad fram till studiens slut. Slutpunkten definierades som ej progress om kröken ökade med 6° eller mindre vid skelettmognad (mindre än 1 cm längdtillväxt under 6 månader) eller progress om kröken ökade mer än 6° före skelettmognad. I delarbete IV användes data från RCT-studien till en longitudinell kohortanalys för att utveckla och validera en prognostisk modell med Cox Proportional Hazards överlevnadsanalys.  

Resultat: Delstudie I visade nedsatt postural stabilitet hos patienter med AIS jämfört med CON genom ökat postural svaj, även framåt-bakåt samt i sidled med tyngdpunkten förskjuten mot hälarna. I RCT-studien deltog 135 individer med en medelålder på 12,7 år (SD1.4) och en Cobb vinkel på 31° (SD5.3). Vid 6 månader fanns hög följsamhet (72-95%) och motivation (65-92%) till behandlingen, med den högsta andelen i NB-gruppen. Både SSE- och PA-grupperna ökade sin totala nivå av fysisk aktivitet per vecka jämfört med NB-gruppen. Vid studiens slut hade SSE-gruppen en högre andel av måttliga problem när det gällde att kunna röra sig och utföra vanliga aktiviteter jämfört med NB- och PA-grupperna, utan andra mellangruppsskillnader. Adekvat följsamhet till behandlingen kvarstod men var bättre i NB- och PA-grupperna jämfört med SSE-gruppen (50-89%). Följsamheten ökade med högre grad av egenförmåga och motivation vilket var tydligare vid studiens slut jämfört med vid 6 månader. Den prognostiska modellen (n=127) för kurvprogess visade acceptabel diskrimineringsförmåga (0.791) där riskfaktorer var skelettomognad, större ryggradskrökning samt försämrad uppfattning om ryggens utseende. Modellen justerades för behandling då nattkorsett minskade risken för ökad ryggradskrökning.  

Konklusion: Patienter med AIS hade nedsatt postural stabilitet med en tyngdpunktsförskjutning mot hälarna. Utvärderingen av behandlingen visade få skillnader mellan grupperna vid studiens slut. SSE-gruppen hade fler med måttliga problem att röra sig och utföra aktiviteter jämfört med övriga grupper. Följsamheten till behandlingen var adekvat vid slutpunkten men var bättre i NB- och PA-grupperna. Högre motivation och egenförmåga ökade långsiktigt följsamheten till behandlingen. Behandlingsalternativen skulle kunna ses som första alternativ efter kliniskt beslut om behandling. Att inkludera självrapport om ryggens utseende som riskfaktor i den prognostiska modellen kan vara kliniskt viktigt för att förutsäga vilka patienter som riskerar progress av kröken.   

Place, publisher, year, edition, pages
Linköping: Linköping University Electronic Press, 2024. p. 149
Series
Linköping University Medical Dissertations, ISSN 0345-0082 ; 1876
Keywords
Idiopathic Scoliosis, Postural stability, Spinal Appearance, Health-Related Quality of Life, Adherence, Physical Activity, Brace, Scoliosis-Specific Exercise, Prognosis, Survival Models
National Category
Physiotherapy
Identifiers
urn:nbn:se:liu:diva-203011 (URN)10.3384/9789180753548 (DOI)9789180753531 (ISBN)9789180753548 (ISBN)
Public defence
2024-05-31, Hasselqvistsalen, Building 511/001, Campus US, Linköping, 09:00 (Swedish)
Opponent
Supervisors
Available from: 2024-04-24 Created: 2024-04-24 Last updated: 2025-04-22Bibliographically approved

Open Access in DiVA

fulltext(854 kB)44 downloads
File information
File name FULLTEXT01.pdfFile size 854 kBChecksum SHA-512
d9a85b947e8398f0941ba54de4136adf4a1a9c16a4b9f0667561561b943b01d934af37e17fab163fe3a2b75f927373379d393b3928429493a03c75ea58cd3d59
Type fulltextMimetype application/pdf

Other links

Publisher's full textPubMed

Search in DiVA

By author/editor
Dufvenberg, MarleneCharalampidis, AnastasiosÖberg, BirgittaTropp, HansWezenberg, DaphneHedevik, HenrikGerdhem, PaulAbbott, Allan
By organisation
Faculty of Medicine and Health SciencesDivision of Prevention, Rehabilitation and Community MedicineDepartment of Orthopaedics in LinköpingDivision of Surgery, Orthopedics and OncologyCenter for Medical Image Science and Visualization (CMIV)
In the same journal
Acta Orthopaedica
Cancer and Oncology

Search outside of DiVA

GoogleGoogle Scholar
Total: 44 downloads
The number of downloads is the sum of all downloads of full texts. It may include eg previous versions that are now no longer available

doi
pubmed
urn-nbn

Altmetric score

doi
pubmed
urn-nbn
Total: 334 hits
CiteExportLink to record
Permanent link

Direct link
Cite
Citation style
  • apa
  • ieee
  • modern-language-association-8th-edition
  • vancouver
  • Other style
More styles
Language
  • de-DE
  • en-GB
  • en-US
  • fi-FI
  • nn-NO
  • nn-NB
  • sv-SE
  • Other locale
More languages
Output format
  • html
  • text
  • asciidoc
  • rtf