Download PDF
Thromb Haemost 2021; 121(10): 1367-1375
DOI: 10.1055/s-0041-1725199
Stroke, Systemic or Venous Thromboembolism

Validation of Outcome Instruments for Pediatric Postthrombotic Syndrome: Introducing the Peds-VEINES-QOL, a New Health-Related Quality of Life Instrument

Julie Smith
1   Hemophilia and Thrombosis Center, Department of Pediatrics, Children's Hospital Colorado, University of Colorado Anschutz Medical Campus, Aurora, Colorado, United States
,
Dianne Thornhill
1   Hemophilia and Thrombosis Center, Department of Pediatrics, Children's Hospital Colorado, University of Colorado Anschutz Medical Campus, Aurora, Colorado, United States
,
Neil A. Goldenberg
2   Department of Pediatrics and Medicine, Johns Hopkins University School of Medicine, Baltimore, Maryland, United States
3   Johns Hopkins All Children's Institute for Clinical and Translational Research, St. Petersburg, Florida, United States
,
Leonardo Brandão 
4   The Hospital for Sick Children (SickKids), University of Toronto, Toronto, Canada
,
Rhonda Knapp-Clevenger
5   University of Rochester Medical Center, School of Nursing, University of Rochester, Rochester, New York, United States
,
Beth Boulden Warren
1   Hemophilia and Thrombosis Center, Department of Pediatrics, Children's Hospital Colorado, University of Colorado Anschutz Medical Campus, Aurora, Colorado, United States
,
Lillian Sung
4   The Hospital for Sick Children (SickKids), University of Toronto, Toronto, Canada
6   Peter Gilgan Centre for Research and Learning, The Hospital for Sick Children, Toronto, Canada
,
Susan R. Kahn
7   Department of Medicine, Lady Davis Institute, McGill University, Montreal, Quebec, Canada
,
Marilyn Manco-Johnson
1   Hemophilia and Thrombosis Center, Department of Pediatrics, Children's Hospital Colorado, University of Colorado Anschutz Medical Campus, Aurora, Colorado, United States
› Author Affiliations Funding This study was supported by the NIH U34 5U34HL114988, which included salary support for N.A.G. and M.M.J. J.S., D.T., B.B.W., N.G., and M.M.J. received salary support from the Health Resources and Services Administration (HRSA) of the U.S. Department of Health and Human Services (HHS), Maternal Child Health Bureau (MCHB) (2H30MC24049). S.R.K. is a Tier 1 Canada Research Chair holder, and an investigator of the CanVECTOR Network, which receives grant funding from the Canadian Institutes of Health Research (Funding Reference: CDT-142654).
› Further Information
    Permissions and Reprints

Abstract

Background There is need for validated outcome measures for postthrombotic syndrome (PTS) following pediatric venous thromboembolism (VTE), with a focus on quality of life (QoL).

Aims This article assesses reliability and validity of two PTS and two QoL scales for children following lower extremity VTE.

Methods Pediatric patients following lower extremity VTE were recruited from three thrombosis clinics. The Manco–Johnson (MJ) and the modified Villalta (MV) PTS scales were compared with each other and with the generic pediatric health-related QoL, PedsQL, and a newly developed pediatric venous-specific QoL, the Peds-VEINES-QOL.

Results Eighty children following VTE and 60 healthy control children were enrolled. Internal consistency measured by Cronbach's α was high for the two QoL scales, and moderate for the two PTS scales. Inter-rater reliability using intraclass correlation coefficients was moderate to high for the MJ, MV, and Peds-VEINES-QOL, and moderate for the PedsQL. Evidence of high internal consistency by Cronbach's α coefficients, and moderate to high interitem correlations support the premise that a single construct was measured by each instrument. Correlations between the four instruments indicate convergent validity.

Conclusion The MJ and MV scales detect similar outcomes in children following VTE. As used, the MJ is slightly more sensitive to QoL because a positive diagnosis requires pain which is the leading factor in reduced QoL following deep vein thrombosis. When using the MV, a requirement for pain or abnormal use to diagnose PTS would make the MV a better predictor of QoL.

Keywords

Manco–Johnson instrument - modified Villalta instrument - Peds-VEINES-QOL - PedsQL - pediatric VTE outcomes

Supplementary Material



Publication History

Received: 09 September 2020

Accepted: 23 January 2021

Article published online:
11 March 2021

© 2021. Thieme. All rights reserved.

Georg Thieme Verlag KG
Rüdigerstraße 14, 70469 Stuttgart, Germany

 

  • References

  • 1 Goldenberg NA, Brandão L, Journeycake J. et al; Perinatal And Paediatric Haemostasis Subcommittee Of The Scientific And Standardization Committee Of The International Society On Thrombosis And Haemostasis. Definition of post-thrombotic syndrome following lower extremity deep venous thrombosis and standardization of outcome measurement in pediatric clinical investigations. J Thromb Haemost 2012; 10 (03) 477-480
    CrossrefPubMedGoogle Scholar
  • 2 Kachroo S, Boyd Bookhart BK. et al. Quality of life and economic costs associated with postthrombotic syndrome. Am J Health-Syst Pharmacy 2012; 69 (07) 567-572
    CrossrefPubMedGoogle Scholar
  • 3 Goldenberg NA, Donadini MP, Kahn SR. et al. Post-thrombotic syndrome in children: a systematic review of frequency of occurrence, validity of outcome measures, and prognostic factors. Haematologica 2010; 95 (11) 1952-1959
    CrossrefPubMedGoogle Scholar
  • 4 Kuhle S, Koloshuk B, Marzinotto V. et al. A cross-sectional study evaluating post-thrombotic syndrome in children. Thromb Res 2003; 111 (4-5): 227-233
    CrossrefPubMedGoogle Scholar
  • 5 Raffini L, Davenport J, Bevilacqua L, Iosifescu S. Comparison of 3 postthrombotic syndrome assessment scales demonstrates significant variability in children and adolescents with deep vein thrombosis. J Pediatr Hematol Oncol 2015; 37 (08) 611-615
    CrossrefPubMedGoogle Scholar
  • 6 Goldenberg NA, Durham JD, Knapp-Clevenger R, Manco-Johnson MJ. A thrombolytic regimen for high-risk deep venous thrombosis may substantially reduce the risk of postthrombotic syndrome in children. Blood 2007; 110 (01) 45-53
    CrossrefPubMedGoogle Scholar
  • 7 Kahn SR, Lamping DL, Ducruet T. et al; VETO Study investigators. VEINES-QOL/Sym questionnaire was a reliable and valid disease-specific quality of life measure for deep venous thrombosis. J Clin Epidemiol 2006; 59 (10) 1049-1056
    PubMedGoogle Scholar
  • 8 Varni JW, Seid M, Kurtin PS. PedsQL 4.0: reliability and validity of the Pediatric Quality of Life Inventory version 4.0 generic core scales in healthy and patient populations. Med Care 2001; 39 (08) 800-812
    CrossrefPubMedGoogle Scholar
  • 9 Manco-Johnson MJ. Postthrombotic syndrome in children. Acta Haematol 2006; 115 (3-4): 207-213
    CrossrefPubMedGoogle Scholar
  • 10 Wong DL, Baker CM. Pain in children: comparison of assessment scales. Pediatr Nurs 1988; 14 (01) 9-17
    Google Scholar
  • 11 Hicks CL, von Baeyer CL, Spafford PA, van Korlaar I, Goodenough B. The Faces Pain Scale-Revised: toward a common metric in pediatric pain measurement. Pain 2001; 93 (02) 173-183
    CrossrefPubMedGoogle Scholar
  • 12 Garra G, Singer AJ, Domingo A, Thode Jr HC. The Wong-Baker pain FACES scale measures pain, not fear. Pediatr Emerg Care 2013; 29 (01) 17-20
    CrossrefPubMedGoogle Scholar
  • 13 von Baeyer CL. Children's self-reports of pain intensity: scale selection, limitations and interpretation. Pain Res Manag 2006; 11 (03) 157-162
    CrossrefPubMedGoogle Scholar
  • 14 Chambers CT, Giesbrecht K, Craig KD, Bennett SM, Huntsman E. A comparison of faces scales for the measurement of pediatric pain: children's and parents' ratings. Pain 1999; 83 (01) 25-35
    CrossrefPubMedGoogle Scholar
  • 15 Eklöf B, Rutherford RB, Bergan JJ. et al; American Venous Forum International Ad Hoc Committee for Revision of the CEAP Classification. Revision of the CEAP classification for chronic venous disorders: consensus statement. J Vasc Surg 2004; 40 (06) 1248-1252
    CrossrefPubMedGoogle Scholar
  • 16 Goldenberg NA, Branchford B, Wang M, Ray Jr C, Durham JD, Manco-Johnson MJ. Percutaneous mechanical and pharmacomechanical thrombolysis for occlusive deep vein thrombosis of the proximal limb in adolescent subjects: findings from an institution-based prospective inception cohort study of pediatric venous thromboembolism. J Vasc Interv Radiol 2011; 22 (02) 121-132
    CrossrefPubMedGoogle Scholar
  • 17 Wik HS, Enden TR, Ghanima W, Engeseth M, Kahn SR, Sandset PM. Diagnostic scales for the post-thrombotic syndrome. Thromb Res 2018; 164: 110-115
    CrossrefPubMedGoogle Scholar
  • 18 Varni JW, Burwinkle TM, Seid M. The PedsQL as a pediatric patient-reported outcome: reliability and validity of the PedsQL Measurement Model in 25,000 children. Expert Rev Pharmacoecon Outcomes Res 2005; 5 (06) 705-719
    CrossrefPubMedGoogle Scholar
  • 19 Kahn SR, Hirsch A, Shrier I. Effect of postthrombotic syndrome on health-related quality of life after deep venous thrombosis. Arch Intern Med 2002; 162 (10) 1144-1148
    CrossrefPubMedGoogle Scholar
  • 20 Kahn SR, Elman EA, Bornais C, Blostein M, Wells PS. Post-thrombotic syndrome, functional disability and quality of life after upper extremity deep venous thrombosis in adults. Thromb Haemost 2005; 93 (03) 499-502
    Article in Thieme ConnectPubMedGoogle Scholar
  • 21 Lamping DL, Schroter S, Kurz X, Kahn SR, Abenhaim L. Evaluation of outcomes in chronic venous disorders of the leg: development of a scientifically rigorous, patient-reported measure of symptoms and quality of life. J Vasc Surg 2003; 37 (02) 410-419
    CrossrefPubMedGoogle Scholar
  • 22 Goldenberg NA, Abshire T, Blatchford PJ. et al; Kids-DOTT Trial Investigators. Multicenter randomized controlled trial on Duration of Therapy for Thrombosis in Children and Young Adults (the Kids-DOTT trial): pilot/feasibility phase findings. J Thromb Haemost 2015; 13 (09) 1597-1605
    CrossrefPubMedGoogle Scholar
  • 23 Rothman M, Burke L, Erickson P, Leidy NK, Patrick DL, Petrie CD. Use of existing patient-reported outcome (PRO) instruments and their modification: the ISPOR good research practices for evaluating and documenting content validity for the use of existing instruments and their modification PRO task force report. Value Health 2009; 12 (08) 1075-1083
    CrossrefPubMedGoogle Scholar
  • 24 DeVellis RF. Scale Development: Theory and Applications. 4th ed.. Los Angeles: SAGE Publications, Inc.; 2017
    Google Scholar
  • 25 Le May S, Ballard A, Khadra C. et al. Comparison of the psychometric properties of 3 pain scales used in the pediatric emergency department: Visual Analogue Scale, Faces Pain Scale-Revised, and Colour Analogue Scale. Pain 2018; 159 (08) 1508-1517
    CrossrefPubMedGoogle Scholar
  • 26 Avila ML, Brandão LR, Williams S. et al. Pediatric post-thrombotic syndrome in children: toward the development of a new diagnostic and evaluative measurement tool. Thromb Res 2016; 144: 184-191
    CrossrefPubMedGoogle Scholar