Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
. 2022 Feb 5;23(1):121.
doi: 10.1186/s12891-022-05070-w.

Test-retest reliability for performance-based outcome measures among individuals with arthrogryposis multiplex congenita

Jaclyn Megan Sions  1 , Maureen Donohoe  2 , Emma Haldane Beisheim-Ryan  3   4 , Ryan Todd Pohlig  5 , Tracy Michele Shank  3 , Louise Reid Nichols  6
Affiliations

Test-retest reliability for performance-based outcome measures among individuals with arthrogryposis multiplex congenita

Jaclyn Megan Sions et al. BMC Musculoskelet Disord. .

Abstract

Background: Most individuals with arthrogryposis multiplex congenita, a rare condition characterized by joint contractures in ≥ 2 body regions, have foot and ankle involvement leading to compromised gait and balance. The purpose of this study was to establish between-days, test-retest reliability for performance-based outcome measures evaluating gait and balance, i.e., the 10-m Walk Test, Figure-of-8 Walk Test, 360-degree Turn Test, and modified Four Square Step Test, among adolescents and adults with arthrogryposis multiplex congenita.

Methods: This reliability study included ambulatory participants, aged 10 to 50 years, with a medical diagnosis of arthrogryposis multiplex congenita. Participants completed performance-based measures, in a randomized order, on two separate occasions. Intraclass correlation coefficients with 95% confidence intervals and minimal detectable changes at the 90% and 95% confidence level were calculated.

Results: Participants included 38 community-ambulators with a median of 13 out of 14 upper and lower joint regions affected. Intraclass correlation coefficient point estimates and 95% confidence intervals ranged from .85-.97 and .70-.98, respectively. Minimal detectable changes were 10 to 39% of sample means and were largest for the modified Four Square Step Test.

Conclusions: Among individuals with arthrogryposis, gait speed per the 10-m Walk Test, as well as non-linear walking and dynamic balance assessment per the Figure-of-8 Walk and 360 Degree Turn Tests, have adequate test-retest reliability enabling evaluation of individual patient changes. Changes in groups of ambulatory individuals with arthrogryposis multiplex congenita may be reliably evaluated with all of the studied outcome measures.

Keywords: Arthrogryposis; Clubfoot; Gait; Postural balance; Walking speed.

PubMed Disclaimer

Conflict of interest statement

LRN is an educational speaker for Smith and Nephew and Orthopediatrics. All other authors declare they have no competing interests.

Figures

Fig. 1
Fig. 1
Study Timeline
Fig. 2
Fig. 2
Standardized Procedures for Performance-Based Outcome Measures
See this image and copyright information in PMC

References

    1. Dahan-Oliel N, Cachecho S, Barnes D, Bedard T, Davison AM, Dieterich K, et al. International multidisciplinary collaboration toward an annotated definition of arthrogryposis multiplex congenita. Am J Med Genet C Semin Med Genet. 2019;181(3):288–299. doi: 10.1002/ajmg.c.31721. - DOI - PMC - PubMed
    1. Mennen U, van Heest A, Ezaki MB, Tonkin M, Gericke G. Arthrogryposis multiplex congenita. J Hand Surg Br. 2005;30(5):468–474. doi: 10.1016/j.jhsb.2005.06.004. - DOI - PubMed
    1. Dahan-Oliel N, van Bosse HJP, Bedard T, Darsaklis VB, Hall JG, Hamdy RC. Research platform for children with arthrogryposis multiplex congenita: Findings from the pilot registry. Am J Med Genet C Semin Med Genet. 2019;181(3):427–435. doi: 10.1002/ajmg.c.31724. - DOI - PubMed
    1. Eriksson M, Villard L, Bartonek A. Walking, orthoses and physical effort in a Swedish population with arthrogryposis. J Child Orthop. 2014;8(4):305–312. doi: 10.1007/s11832-014-0597-9. - DOI - PMC - PubMed
    1. Hall JG. Arthrogryposis (multiple congenital contractures): diagnostic approach to etiology, classification, genetics, and general principles. Eur J Med Genet. 2014;57(8):464–472. doi: 10.1002/ajmg.c.31716. - DOI - PubMed

LinkOut - more resources