Outcomes and outcome measurement instruments in lower-limb lengthening surgery: a scoping review to inform core outcome set development
DOI:
https://doi.org/10.2340/17453674.2024.42488Keywords:
Bone, Implants, Paediatric orthopaedicsAbstract
Background and purpose: The heterogeneous outcomes used in lower-limb lengthening surgery (LLLS) complicate evidence synthesis, weakening systematic reviews and clinical recommendations, and reducing research impact. This scoping review maps the outcomes and outcome measurement instruments (OMIs) used in LLLS.
Methods: This pre-registered review systematically identified studies reporting outcomes in adults or children who underwent LLLS. Outcomes and OMIs were extracted verbatim, and experts grouped outcome terms under headings using the COMET taxonomy.
Results: The search found 5,308 unique hits, including 149 studies from 2020–2024. They reported 2,939 verbatim outcomes, which were consolidated into 92 outcome headings and 27 subheadings. “Life impact” accounted for 13%, while “Clinical outcomes” represented 83% of all outcomes. Among the clinical outcomes, “Musculoskeletal and connective tissue” was the most reported outcome domain (68% of all outcomes). The most frequently reported outcomes were “Lengthening,” “Bone healing,” “Range of motion,” “Limb alignment”, and “Complications.”
Conclusion: Outcomes reported for people undergoing LLLS are heterogeneous and vary widely in the definitions and measurement tools used to collect them. Outcomes likely to be important to patients (life impact outcomes), such as quality of life and measures of physical function, are rarely reported.
Downloads
References
Birch J G. A brief history of limb lengthening. J Pediatr Orthop 2017; 37 Suppl 2(6): S1-8. doi: 10.1097/BPO.0000000000001021. DOI: https://doi.org/10.1097/BPO.0000000000001021
Mittal A, Allahabadi S, Jayaram R, Nalluri A, Callahan M, Sabharwal S. Trends and practices in limb lengthening: an 11-year US database study. Strategies Trauma Limb Reconstr 2023; 18(1): 21-31. doi: 10.5005/jp-journals-10080-1574. DOI: https://doi.org/10.5005/jp-journals-10080-1574
Horn J, Grimsrud Ø, Dagsgard A H, Huhnstock S, Steen H. Femoral lengthening with a motorized intramedullary nail. Acta Orthop 2015; 86(2): 248-56. doi: 10.3109/17453674.2014.960647. DOI: https://doi.org/10.3109/17453674.2014.960647
Hlukha L P, Sax O C, Kowalewski K A, Bains S S, Dubin J, Herzenberg J E, et al. Chronic knee pain following infrapatellar/suprapatellar magnetic intramedullary lengthening nails versus external fixators in limb length discrepancy: a retrospective review. J Orthop 2023; 51:7-11. doi: 10.1016/j.jor.2023.11.071. DOI: https://doi.org/10.1016/j.jor.2023.11.071
Kirkham J J, Williamson P. Core outcome sets in medical research. BMJ Medicine 2022; 1(1): e000284. doi: 10.1136/bmjmed-2022-000284. DOI: https://doi.org/10.1136/bmjmed-2022-000284
Williamson P R, Altman D G, Bagley H, Barnes K L, Blazeby J M, Brookes S T, et al. The COMET Handbook: version 1.0. Trials 2017; 18(Suppl 3): 280. doi: 10.1186/s13063-017-1978-4. DOI: https://doi.org/10.1186/s13063-017-1978-4
Kim S J, Pierce W, Sabharwal S. The etiology of short stature affects the clinical outcome of lower limb lengthening using external fixation: a systematic review of 18 trials involving 547 patients. Acta Orthop 2014; 85(2): 181. doi: 10.3109/17453674.2014.899856. DOI: https://doi.org/10.3109/17453674.2014.899856
Frost M W, Rahbek O, Traerup J, Ceccotti A A, Kold S. Systematic review of complications with externally controlled motorized intramedullary bone lengthening nails (FITBONE and PRECICE) in 983 segments. Acta Orthop 2021; 92(1): 120-7. doi: 10.1080/17453674.2020.1835321. DOI: https://doi.org/10.1080/17453674.2020.1835321
Sheridan G A, Fragomen A T, Rozbruch S R. Integrated limb lengthening is superior to classical limb lengthening: a systematic review and meta-analysis of the literature. J Am Acad Orthop Surg Glob Res Rev. 2020; 4(6). doi: 10.5435/JAAOSGLOBAL-D-20-00054 DOI: https://doi.org/10.5435/JAAOSGlobal-D-20-00054
Axelrod D, Rubinger L, Shah A, Guy P, Johal H. How should we lengthen post-traumatic limb defects? A systematic review and comparison of motorized lengthening systems, combined internal and external fixation and external fixation alone. Eur J Orthop SurgTraumatol 2021; 31(6): 1015-22. doi: 10.1007/s00590-020-02831-y. DOI: https://doi.org/10.1007/s00590-020-02831-y
Frost M W, Rahbek O, Iobst C, Bafor A, Duncan M, Kold S. Complications and risk factors of intramedullary bone lengthening nails: a retrospective multicenter cohort study of 314 FITBONE and PRECICE nails. Acta Orthop 2023; 94: 51. doi: 10.2340/17453674.2023.8479. DOI: https://doi.org/10.2340/17453674.2023.8479
Wang A, Menon R, Li T, Harris L, Harris I A, Naylor J, et al. Has the degree of outcome reporting bias in surgical randomized trials changed? A meta-regression analysis. ANZ J Surg 2023; 93(1-2): 76-82. doi: 10.1111/ans.18273. DOI: https://doi.org/10.1111/ans.18273
Menon R, Wang A, Chamberlain K, Harris L, Li T, Harris I A, et al. Has the reporting of patient-important outcomes improved in surgical trials? A meta-epidemiological study. ANZ J Surg 2021; 91(10): 2014-20. doi: 10.1111/ans.16922. DOI: https://doi.org/10.1111/ans.16922
Saddawi-Konefka D, Kim H M, Chung K C. A systematic review of outcomes and complications of reconstruction and amputation for Type IIIB and IIIC fractures of the tibia. Plast Reconstr Surg 2008; 122(6): 1796-1805. doi: 10.1097/PRS.0b013e31818d69c3. DOI: https://doi.org/10.1097/PRS.0b013e31818d69c3
Tricco A C, Lillie E, Zarin W, O’Brien K K, Colquhoun H, Levac D, et al. PRISMA extension for scoping reviews (PRISMA-ScR): checklist and explanation. Ann Intern Med 2018; 169(7): 467-73. doi: 10.7326/M18-0850. DOI: https://doi.org/10.7326/M18-0850
Peters M D, Godfrey C, McInerney P, Munn Z, Tricco A C, Khalil H. Scoping reviews. JBI Manual for Evidence Synthesis. JBI; 2024. doi: 10.46658/JBIMES-24-09. DOI: https://doi.org/10.46658/JBIMES-24-09
Ouzzani M, Hammady H, Fedorowicz Z, Elmagarmid A. Rayyan—a web and mobile app for systematic reviews. Syst Rev 2016; 5(1): 210. doi: 10.1186/s13643-016-0384-4. DOI: https://doi.org/10.1186/s13643-016-0384-4
Harris P A, Taylor R, Minor B L, Elliott V, Fernandez M, O’Neal L, et al. The REDCap consortium: building an international community of software platform partners. J Biomed Inform 2019; 95: 103208. doi: 10.1016/j.jbi.2019.103208. DOI: https://doi.org/10.1016/j.jbi.2019.103208
Harris P A, Taylor R, Thielke R, Payne J, Gonzalez N, Conde J G. Research electronic data capture (REDCap): a metadata-driven methodology and workflow process for providing translational research informatics support. J Biomed Inform 2009; 42(2): 377-81. doi: 10.1016/j.jbi.2008.08.010. DOI: https://doi.org/10.1016/j.jbi.2008.08.010
Dodd S, Clarke M, Becker L, Mavergames C, Fish R, Williamson P R. A taxonomy has been developed for outcomes in medical research to help improve knowledge discovery. J Clin Epidemiol 2018; 96: 84-92. doi: 10.1016/j.jclinepi.2017.12.020. DOI: https://doi.org/10.1016/j.jclinepi.2017.12.020
Boers M, Kirwan J R, Wells G, Beaton D, Gossec L, D’Agostino M A, et al. Developing core outcome measurement sets for clinical trials: OMERACT filter 2.0. J Clin Epidemiol 2014; 67(7): 745-53. doi: 10.1016/j.jclinepi.2013.11.013. DOI: https://doi.org/10.1016/j.jclinepi.2013.11.013
International Classification of Functioning, Disability and Health (ICF) [Internet]. [cited 2022 Sep 29]. Available from: https://icd.who.int/dev11/l-icf/en#
Beaton D, Maxwell L, Grosskleg S, Shea B, Tugwell B. Chapter 4: Developing core domain sets [Internet]. The OMERACT Handbook Version 21 [updated April 2021] OMERACT. [cited 2024 Jun 26]. Available from: https://omeract.org/handbook/
Page M J, Huang H, Verhagen A P, Gagnier J J, Buchbinder R. Outcome reporting in randomized trials for shoulder disorders: literature review to inform the development of a core outcome set. Arthritis Care Res (Hoboken) 2018; 70(2): 252-9. doi: 10.1002/acr.23254. DOI: https://doi.org/10.1002/acr.23254
Flourish | Data Visualization & Storytelling [Internet]. [cited 2024 Jul 1]. Available from: https://app.flourish.studio/login
Aquilina A L, Claireaux H, Aquilina C O, Tutton E, Fitzpatrick R, Costa M L, et al. What outcomes have been reported on patients following open lower limb fracture, and how have they been measured? Bone Joint Res 2023; 12(2): 138-46. doi: 10.1302/2046-3758.122.BJR-2022-0116.R1. DOI: https://doi.org/10.1302/2046-3758.122.BJR-2022-0116.R1
Kottner J, Beaton D, Clarke M, Dodd S, Kirkham J, Lange T, et al. Core outcome set developers should consider and specify the level of granularity of outcome domains. J Clin Epidemiol 2024; 169. doi: 10.1016/j.jclinepi.2024.111307. DOI: https://doi.org/10.1016/j.jclinepi.2024.111307
Yalcinkaya A, Rahbek O, Tirta M, Rathleff M S, Iobst C, Kold S. Outcomes and outcome measures to inform the development of a core outcome set for lower-limb lengthening surgery: a scoping review protocol. Open Science Framework; 2022. doi: 10.17605/OSF.IO/A2D65.
Haddaway N R, Page M J, Pritchard C C, McGuinness L A. PRISMA2020: An R package and Shiny app for producing PRISMA 2020-compliant flow diagrams, with interactivity for optimised digital transparency and open synthesis. Campbell Syst Rev 2022; 18(2): e1230. doi: 10.1002/cl2.1230. DOI: https://doi.org/10.1002/cl2.1230
Machielsen A J H M, Iqbal N, Kimman M L, Sahnan K, Adegbola S O, Kane G, et al. Heterogeneity in outcome selection, definition and measurement in studies assessing the treatment of cryptoglandular anal fistula: findings from a systematic review. Tech Coloproctol 2021; 25(7): 761. doi: 10.1007/s10151-021-02452-5. DOI: https://doi.org/10.1007/s10151-021-02452-5
Paley D, Catagni M A, Argnani F, Villa A, Battista Benedetti G, Cattaneo R. Ilizarov treatment of tibial nonunions with bone loss. Clin Orthop Relat Res 1989; 241(241): 146-65. doi: 10.1097/00003086-198904000-00017. DOI: https://doi.org/10.1097/00003086-198904000-00017
Fabricant P, Borst E, Green S, Marx R, Fragomen A, Rozbruch S R. Validation of a modified Scoliosis Research Society instrument for patients with limb deformity: The limb deformity-Scoliosis Research Society (LD-SRS) score. J Limb Lengthening Reconstr. 2016; 2(2): 86. doi: 10.4103/2455-3719.190710. DOI: https://doi.org/10.4103/2455-3719.190710
Varni J W, Seid M, Kurtin P S. PedsQLTM 4.0: Reliability and validity of the Pediatric Quality of Life InventoryTM Version 4.0 generic core scales in healthy and patient populations. Med Care 2001; 39(8). doi: 10.1097/00005650-200108000-00006. DOI: https://doi.org/10.1097/00005650-200108000-00006
Iliadis A D, Palloni V, Wright J, Goodier D, Calder P. Pediatric lower limb lengthening using the PRECICE nail: our experience with 50 cases. J Pediatr Orthop 2021; 41(1): e44-9. doi: 10.1097/BPO.0000000000001672 DOI: https://doi.org/10.1097/BPO.0000000000001672
Paley D. Problems, obstacles, and complications of limb lengthening by the Ilizarov technique. Clin Orthop Relat Res 1990; 250(1): 81-104. doi: 10.1097/00003086-199001000-00011. DOI: https://doi.org/10.1097/00003086-199001000-00011
Lascombes P, Popkov D, Huber H, Haumont T, Journeau P. Classification of complications after progressive long bone lengthening: proposal for a new classification. Orthop Traumatol Surg Res 2012; 98(6): 629-37. doi: 10.1016/j.otsr.2012.05.010. DOI: https://doi.org/10.1016/j.otsr.2012.05.010
Swiontkowski M F, Buckwalter J A, Keller R B, Haralson. The outcomes movement in orthopaedic surgery: where we are and where we should go. J Bone Joint Surg Am 1999; 81(5): 732-40. doi: 10.2106/00004623-199905000-00016. DOI: https://doi.org/10.2106/00004623-199905000-00016
Antonios T, Barker A, Ibrahim I, Scarsbrook C, Smitham P J, David Goodier W, et al. A systematic review of patient-reported outcome measures used in circular frame fixation. Strategies Trauma Limb Reconstr 2019; 14(1): 34. doi: 10.5005/jp-journals-10080-1413. DOI: https://doi.org/10.5005/jp-journals-10080-1413
Kim S J, Balce G C, Agashe M V, Song S H, Song H R. Is bilateral lower limb lengthening appropriate for achondroplasia?: midterm analysis of the complications and quality of life. Clin Orthop Relat Res 2012; 470(2): 616-21. doi: 10.1007/s11999-011-1983-y. DOI: https://doi.org/10.1007/s11999-011-1983-y
Bafor A, Iobst C A. what’s new in limb lengthening and deformity correction. J Bone Joint Surg 2022; 104(16): 1419-25. doi: 10.2106/JBJS.22.00398. DOI: https://doi.org/10.2106/JBJS.22.00398
Modin M, Ramos T, Stomberg M W. Postoperative impact of daily life after primary treatment of proximal/distal tibia fracture with Ilizarov external fixation. J Clin Nurs 2009; 18(24): 3498-506. doi: 10.1111/j.1365-2702.2009.02859.x. DOI: https://doi.org/10.1111/j.1365-2702.2009.02859.x
Hrutkay J M, Eilert R E. Operative lengthening of the lower extremity and associated psychological aspects: the Children’s Hospital experience. J Pediatr Orthop 1990; 10(3): 373-7. doi: 10.1097/01241398-199005000-00015. DOI: https://doi.org/10.1097/01241398-199005000-00015
World Bank Country and Lending Groups—World Bank Data Help Desk [Internet]. [cited 2024 Jun 26]. Available from: https://datahelpdesk.worldbank.org/knowledgebase/articles/906519-world-bank-country-and-lending-groups
Hafez M, Nicolaou N, Offiah A, Obasohan P, Dixon S, Giles S, et al. How much does paediatric femoral lengthening cost? A cost comparison between magnetic lengthening nails and external fixators. Strategies Trauma Limb Reconstr 2023; 18(1): 16. doi: 10.5005/jp-journals-10080-1573. DOI: https://doi.org/10.5005/jp-journals-10080-1573
Additional Files
Published
How to Cite
License
Copyright (c) 2024 Ali Yalcinkaya, Ole Rahbek, Maria Tirta, Jette Frost Jepsen, Michael Skovdal Rathleff, Christopher Iobst, Søren Kold
This work is licensed under a Creative Commons Attribution 4.0 International License.
