title: PedsQL™ Multidimensional Fatigue Scale in sickle cell disease: feasibility, reliability, and validity.
abstract: BACKGROUND
  Sickle cell disease (SCD) is an inherited blood disorder characterized by a chronic hemolytic anemia that can contribute to fatigue and global cognitive impairment in patients. The study objective was to report on the feasibility, reliability, and validity of the PedsQL™ Multidimensional Fatigue Scale in SCD for pediatric patient self-report ages 5-18 years and parent proxy-report for ages 2-18 years.
  
  
  METHODS
  This was a cross-sectional multi-site study whereby 240 pediatric patients with SCD and 303 parents completed the 18-item PedsQL™ Multidimensional Fatigue Scale. Participants also completed the PedsQL™ 4.0 Generic Core Scales.
  
  
  RESULTS
  The PedsQL™ Multidimensional Fatigue Scale evidenced excellent feasibility, excellent reliability for the Total Scale Scores (patient self-report α = 0.90; parent proxy-report α = 0.95), and acceptable reliability for the three individual scales (patient self-report α = 0.77-0.84; parent proxy-report α = 0.90-0.97). Intercorrelations of the PedsQL™ Multidimensional Fatigue Scale with the PedsQL™ Generic Core Scales were predominantly in the large (≥0.50) range, supporting construct validity. PedsQL™ Multidimensional Fatigue Scale Scores were significantly worse with large effects sizes (≥0.80) for patients with SCD than for a comparison sample of healthy children, supporting known-groups discriminant validity. Confirmatory factor analysis demonstrated an acceptable to excellent model fit in SCD.
  
  
  CONCLUSIONS
  The PedsQL™ Multidimensional Fatigue Scale demonstrated acceptable to excellent measurement properties in SCD. The results demonstrate the relative severity of fatigue symptoms in pediatric patients with SCD, indicating the potential clinical utility of multidimensional assessment of fatigue in patients with SCD in clinical research and practice.
authors.affiliation: Department of Pediatrics, Division of Hematology/Oncology/Bone Marrow Transplantation, Children's Hospital of Wisconsin of the Children's Research Institute, Medical College of Wisconsin, Milwaukee, Wisconsin.
authors.forename: Julie A
authors.initials: JA
authors.lastname: Panepinto
authors.forename: Sylvia
authors.initials: S
authors.lastname: Torres
authors.forename: Cristiane B
authors.initials: CB
authors.lastname: Bendo
authors.forename: Timothy L
authors.initials: TL
authors.lastname: McCavit
authors.forename: Bogdan
authors.initials: B
authors.lastname: Dinu
authors.forename: Sandra
authors.initials: S
authors.lastname: Sherman-Bien
authors.forename: Christy
authors.initials: C
authors.lastname: Bemrich-Stolz
authors.forename: James W
authors.initials: JW
authors.lastname: Varni
mesh_terms: D000293:Adolescent; D000755:Anemia, Sickle Cell / Q000150:complications* / Q000523:psychology; D002648:Child; D002675:Child, Preschool; D003430:Cross-Sectional Studies; D005221:Fatigue / Q000175:diagnosis* / Q000209:etiology / Q000523:psychology; D005240:Feasibility Studies; D006801:Humans; D007223:Infant; D010290:Parents; D011594:Psychometrics / Q000295:instrumentation*; D011788:Quality of Life; D015203:Reproducibility of Results; D057566:Self Report; D012720:Severity of Illness Index; D011795:Surveys and Questionnaires
keywords: PedsQL; children; fatigue; health-related quality of life; patient-reported outcomes; pediatrics; sickle cell disease
references.citation: Clin Gastroenterol Hepatol. 2009 May;7(5):554-61
references.pmid: 19418604
references.citation: Health Psychol. 2006 Sep;25(5):635-42
references.pmid: 17014281
references.citation: Med Care. 2001 Aug;39(8):800-12
references.pmid: 11468499
references.citation: Multivariate Behav Res. 1990 Apr 1;25(2):173-80
references.pmid: 26794479
references.citation: J Pediatr Health Care. 2011 Jul-Aug;25(4):208-15
references.pmid: 21700135
references.citation: Brain Inj. 2012;26(9):1072-83
references.pmid: 22642370
references.citation: Pediatr Diabetes. 2009 Aug;10(5):321-8
references.pmid: 19067887
references.citation: Pediatr Blood Cancer. 2010 Sep;55(3):485-94
references.pmid: 20658620
references.citation: N Engl J Med. 1996 Aug 8;335(6):369-76
references.pmid: 8663884
references.citation: Qual Life Res. 2011 Sep;20(7):1091-102
references.pmid: 21225351
references.citation: Pediatr Blood Cancer. 2013 Aug;60(8):1338-44
references.pmid: 23441057
references.citation: Arch Clin Neuropsychol. 2005 Dec;20(8):1073-85
references.pmid: 16139469
references.citation: J Rheumatol. 2004 Dec;31(12):2494-500
references.pmid: 15570657
references.citation: Qual Life Res. 2008 Aug;17(6):895-913
references.pmid: 18521721
references.citation: Qual Life Res. 1996 Jun;5(3):321-9
references.pmid: 8763800
references.citation: Qual Life Res. 1995 Aug;4(4):293-307
references.pmid: 7550178
references.citation: Med Care. 1994 Jan;32(1):40-66
references.pmid: 8277801
references.citation: Psychol Bull. 1990 Mar;107(2):238-46
references.pmid: 2320703
references.citation: J Pediatr Hematol Oncol. 2008 Sep;30(9):666-73
references.pmid: 18776758
references.citation: J Pediatr. 1996 Jun;128(6):820-8
references.pmid: 8648542
references.citation: J Nurs Scholarsh. 2011 Mar;43(1):22-9
references.pmid: 21342421
references.citation: Qual Life Res. 2011 Sep;20(7):1103-8
references.pmid: 21246290
references.citation: J Pediatr Hematol Oncol. 2011 May;33(4):251-4
references.pmid: 21516020
references.citation: Qual Life Res. 2013 Nov;22(9):2581-94
references.pmid: 23423759
references.citation: Qual Life Res. 1997 Mar;6(2):139-50
references.pmid: 9161114
references.citation: Int J Pediatr Obes. 2010;5(1):34-42
references.pmid: 19593727
references.citation: AJNR Am J Neuroradiol. 2003 Mar;24(3):382-9
references.pmid: 12637286
references.citation: Cancer. 2002 Apr 1;94(7):2090-106
references.pmid: 11932914
references.citation: Int J Behav Med. 2006;13(3):252-8
references.pmid: 17078776
references.citation: J Pediatr Psychol. 2012 Aug;37(7):798-807
references.pmid: 22467881
references.citation: Arch Pediatr Adolesc Med. 2008 May;162(5):469-75
references.pmid: 18458194
references.citation: Qual Life Res. 2012 Mar;21(2):341-57
references.pmid: 21638090
references.citation: N Engl J Med. 1995 May 18;332(20):1317-22
references.pmid: 7715639
references.citation: Pediatrics. 2008 Mar;121(3):e449-57
references.pmid: 18310166
references.citation: Pediatr Blood Cancer. 2010 Jan;54(1):92-7
references.pmid: 19653296
references.citation: Neurology. 2001 Apr 24;56(8):1109-11
references.pmid: 11320190