Abstract
Study design
Cross sectional
Objectives
The purpose of this study is to evaluate the association between thoracic height and health-related quality of life (HRQoL) at skeletal maturity in patients with EOS.
Summary of background data
Current literature suggests a minimum thoracic height of 18 cm to 22 cm to avoid poor pulmonary function and related health outcomes.
Methods
Patients with EOS who reached skeletal maturity from 2005 to 2018 were identified in two registries including 32 centers. Thoracic height from T1 to T12 at skeletal maturity and Early Onset Scoliosis 24 Item Questionnaire (EOSQ-24) scores were collected. The EOSQ-24 domains included HRQoL of patients, parental impact, financial impact and patient and parental satisfaction.
Results
469 patients (mean age: 14.9, female: 77.4%) were identified. 29% patients were of congenital etiology, 20.3% neuromuscular, 13.6% syndromic, 34.8% idiopathic, and 2.3% other. When patients were grouped by thoracic height at skeletal maturity, all EOSQ-24 domains increased after a threshold of 18 cm. When stratified by etiology, the 18 cm cutoff held for patients with congenital, neuromuscular and syndromic EOS. The cutoff for idiopathic EOS was 20 cm. For all patients, after the threshold was met, HRQoL continued to improve with increases in thoracic height at skeletal maturity. A subset of 169 patients for which arm span measurements were available was also identified and their thoracic heights were normalized. When grouped by the percentage of expected thoracic height attained, EOSQ-24 domains increased after a threshold of 80%.
Conclusions
Once 18 cm of actual thoracic height or 80% of expected thoracic height is achieved, HRQoL continues to improve as thoracic height increases in skeletally mature patients with non-idiopathic EOS. Patients with idiopathic EOS had a higher threshold, possibly due to their larger average size and higher care giver expectations for HRQoL.
Level of evidence
Level III.
Similar content being viewed by others
References
Karol LA, Johnston C, Mladenov K, Schochet P, Walters P, Browne RH (2008) Pulmonary function following early thoracic fusion in non-neuromuscular scoliosis. J Bone Jt Surg 90(6):1272–1281. https://doi.org/10.2106/JBJS.G.00184
Pehrsson K, Larsson S, Oden A, Nachemson A (1992) Long-term follow-up of patients with untreated scoliosis. A study of mortality, causes of death, and symptoms. Spine (Phila Pa 1976) 17(9):1091–1096
Branthwaite MA (1986) Cardiorespiratory consequences of unfused idiopathic scoliosis. Br J Dis Chest 80(4):360–369
Scott JC, Morgan TH (1955) The natural history and prognosis of infantile idiopathic scoliosis. J Bone Joint Surg Br 37-B(3):400–413
Campbell RM, Smith MD, Mayes TC et al (2003) The characteristics of thoracic insufficiency syndrome associated with fused ribs and congenital scoliosis. J Bone Joint Surg Am 85-A(3):399–408
Campbell RM Jr, Smith MD, Mayes TC et al (2003) The characteristics of thoracic insufficiency syndrome associated with fused ribs and congenital scoliosis. J Bone Jt Surg Am 85-A(3):399–408
Vitale MG, Matsumoto H, Roye DP Jr et al (2008) Health-related quality of life in children with thoracic insufficiency syndrome. J Pediatr Orthop 28(2):239–243
Dimeglio A (2011) Growth in pediatric orthopaedics. J Pediatr Orthop. 21(4):549–555
Vitale MG, Matsumoto H, Bye MR et al (2008) A retrospective cohort study of pulmonary function, radiographic measures, and quality of life in children with congenital scoliosis: an evaluation of patient outcomes after early spinal fusion. Spine (Phila Pa 1976) 33(11):1242–1249. https://doi.org/10.1097/BRS.0b013e3181714536
Fletcher ND, Bruce RW (2012) Early onset scoliosis: current concepts and controversies. Curr Rev Musculoskelet Med 5(2):102–110. https://doi.org/10.1007/s12178-012-9116-0
Yang S, Andras LM, Redding GJ, Skaggs DL (2016) Early-onset scoliosis: a review of history, current treatment, and future directions. Pediatrics 137(1):e20150709
Hedequist D, Emans J (2007) Congenital scoliosis: a review and update. J Pediatr Orthop 27(1):106–116
Gillingham BL, Fan RA, Akbarnia BA (2006) Early onset idiopathic scoliosis. J Am Acad Orthop Surg 14(2):101–112
Sankar WN, Skaggs DL, Yazici M et al (2011) Lengthening of dual growing rods and the law of diminishing returns. Spine (Phila Pa 1976) 36(10):806–809. https://doi.org/10.1097/BRS.0b013e318214d78f
Grivas TB, Burwell RG, Purdue M, Webb JK, Moulton A (1991) A segmental analysis of thoracic shape in chest radiographs of children. Changes related to spinal level, age, sex, side and significance for lung growth and scoliosis. J Anat 178:21–38
Pehrsson K, Nachemson A, Olofson J, Ström K, Larsson S (1992) Respiratory failure in scoliosis and other thoracic deformities A survey of patients with home oxygen or ventilator therapy in Sweden. Spine (Phila Pa 1976) 17(6):714–718
Matsumoto H, Williams B, Park HY et al (2018) The final 24-item early onset scoliosis questionnaires (EOSQ-24). J Pediatr Orthop 38(3):144–151. https://doi.org/10.1097/BPO.0000000000000799
Capderou A, Berkani M, Becquemin M-H, Zelter M (2011) Reconsidering the arm span–height relationship in patients referred for spirometry. Eur Respir J 37(1):157–163. https://doi.org/10.1183/09031936.00199209
Arriba Munoz A, Domínguez Cajal M, Rueda Caballero C, Labarta Aizpún JI, Mayayo Dehesa E, Ferrández LÁ (2013) Relación talla sentada/talla de pie del nacimiento a la adultez en niños españoles. Arch Argent Pediatr 111(4):309–314. https://doi.org/10.5546/aap.2013.309
Fredriks M, van Buuren S, van Heel WJM, Dijkman-Neerincx RHM, Verloove-Vanhorick SP, Wit JM (2005) Nationwide age references for sitting height, leg length, and sitting height/height ratio, and their diagnostic value for disproportionate growth disorders. Arch Dis Child 90(8):807–812. https://doi.org/10.1136/adc.2004.050799
Dimeglio A, Canavese F (2012) The growing spine: how spinal deformities influence normal spine and thoracic cage growth. Eur Spine J 21(1):64–70. https://doi.org/10.1007/s00586-011-1983-3
Canavese F, Dimeglio A (2013) Normal and abnormal spine and thoracic cage development. World J Orthop 4(4):167–174. https://doi.org/10.5312/wjo.v4.i4.167
O’Brien M, Kulklo T, Blanke K, Lenke L (2008) Spinal deformity study group radiographic measurement manual. Medtronic Sofamor Danek USA, Inc, Memphis
Funding
This work was conducted without the support of additional funding.
Author information
Authors and Affiliations
Consortia
Contributions
BDR: Resources, Conception and design, data interpretation, revision, final approval. MES: Data analysis, data interpretation, drafting manuscript, revising manuscript, final approval. HM: Conception and design, Data analysis, data interpretation, final approval. SG: Resources, Manuscript revision, final approval. GR: Manuscript revision, final approval. AS: Resources, Manuscript revision, final approval. JTS: Resources, Manuscript revision, final approval. PS: Resources, Manuscript revision, final approval. MGV: Resources, Conception and design, supervision, final approval. Children’s Spine Study Group: Data acquisition, Final Approval. Growing Spine Study Group: Data acquisition, final approval.
Corresponding author
Ethics declarations
IRB approval/Research Ethics Committee
This work is approved by the Institutional Review Board at Columbia University (Protocol AAAS0358).
Additional information
Publisher's Note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
Rights and permissions
About this article
Cite this article
Roye, B.D., Simhon, M.E., Matsumoto, H. et al. Bigger is better: larger thoracic height is associated with increased health related quality of life at skeletal maturity. Spine Deform 8, 771–779 (2020). https://doi.org/10.1007/s43390-020-00095-4
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s43390-020-00095-4