TY - JOUR
T1 - Fatigue in patients with spinal muscular atrophy type II and congenital myopathies
T2 - evaluation of the fatigue severity scale
AU - Werlauff, Ulla
AU - Højberg, A
AU - Firla-Holme, R
AU - Steffensen, B F
AU - Vissing, J
PY - 2014/6
Y1 - 2014/6
N2 - PURPOSE: The aim of this study was to evaluate whether the fatigue severity scale (FSS) is an appropriate instrument to assess fatigue in patients with spinal muscular atrophy type II (SMA II) and congenital myopathies (CM).METHODS: FSS and visual analog scale (VAS) were administered to 33 SMA II- and 72 CM patients. The psychometric properties of the FSS were evaluated by means of classical test theories for each of the disease groups. If abnormal fatigue was present in the disease group, the construct of fatigue was evaluated by means of focus group interviews.RESULTS: Fatigue was rare in SMA II patients, but very frequent in patients with CM. The cut-off score designating abnormal fatigue (FSS score ≥ 4) was exceeded by 10% of the SMA II patients in contrast to 76% of the CM patients, of whom 52% suffered from severe fatigue (FSS score ≥ 5). Focus group interviews demonstrated that fatigue had an adverse effect on motor function, level of energy, social relations, and identity, four themes that could be captured by the FSS. The FSS and VAS were strongly correlated in SMA II patients, but only moderately in CM patients. The psychometric properties indicated that the original FSS with nine items measures more than one construct of fatigue, eliminating the first two items improved scale properties.CONCLUSION: This study demonstrates that fatigue is characteristic in patients with CM, but not in patients with SMA II, in whom fatigue does not seem to impact daily life. While fatigue in CM and SMA II can be captured by FSS, omitting the first two items of the scale will improve its properties and content validity, along with comprehension of the scale itself.
AB - PURPOSE: The aim of this study was to evaluate whether the fatigue severity scale (FSS) is an appropriate instrument to assess fatigue in patients with spinal muscular atrophy type II (SMA II) and congenital myopathies (CM).METHODS: FSS and visual analog scale (VAS) were administered to 33 SMA II- and 72 CM patients. The psychometric properties of the FSS were evaluated by means of classical test theories for each of the disease groups. If abnormal fatigue was present in the disease group, the construct of fatigue was evaluated by means of focus group interviews.RESULTS: Fatigue was rare in SMA II patients, but very frequent in patients with CM. The cut-off score designating abnormal fatigue (FSS score ≥ 4) was exceeded by 10% of the SMA II patients in contrast to 76% of the CM patients, of whom 52% suffered from severe fatigue (FSS score ≥ 5). Focus group interviews demonstrated that fatigue had an adverse effect on motor function, level of energy, social relations, and identity, four themes that could be captured by the FSS. The FSS and VAS were strongly correlated in SMA II patients, but only moderately in CM patients. The psychometric properties indicated that the original FSS with nine items measures more than one construct of fatigue, eliminating the first two items improved scale properties.CONCLUSION: This study demonstrates that fatigue is characteristic in patients with CM, but not in patients with SMA II, in whom fatigue does not seem to impact daily life. While fatigue in CM and SMA II can be captured by FSS, omitting the first two items of the scale will improve its properties and content validity, along with comprehension of the scale itself.
KW - Activities of Daily Living
KW - Adult
KW - Denmark
KW - Fatigue
KW - Female
KW - Focus Groups
KW - Humans
KW - Interpersonal Relations
KW - Interviews as Topic
KW - Male
KW - Neuromuscular Diseases
KW - Psychometrics
KW - Qualitative Research
KW - Reproducibility of Results
KW - Severity of Illness Index
KW - Sickness Impact Profile
KW - Spinal Muscular Atrophies of Childhood
KW - Visual Analog Scale
U2 - 10.1007/s11136-013-0565-8
DO - 10.1007/s11136-013-0565-8
M3 - Journal article
C2 - 24214178
SN - 0962-9343
VL - 23
SP - 1479
EP - 1488
JO - Quality of Life Research
JF - Quality of Life Research
IS - 5
ER -