TY - JOUR
T1 - Severe fatigue and reduced quality of life in children with hereditary motor and sensory neuropathy 1A
AU - Jagersma, Elbrich
AU - Jeukens-Visser, Martine
AU - van Paassen, Barbara W.
AU - Meester-Delver, Anke
AU - Nollet, Frans
PY - 2013
Y1 - 2013
N2 - Severe fatigue and low quality of life are reported by a majority of adult patients with hereditary motor and sensory neuropathy 1A. In children with hereditary motor and sensory neuropathy 1A, the prevalence and impact of fatigue have not been studied yet. In this questionnaire survey, 55 Dutch children (response rate 77%) with genetically confirmed hereditary motor and sensory neuropathy 1A participated (mean age 15 years [standard deviation 2.1]). Prevalence of severe fatigue (based on a cut-off score of the Checklist Individual Strength) was 24%, in contrast to 14% in a Dutch school-based population (P < .05). Almost all quality-of-life scores (measured with the Child Health Questionnaire-Child Form 87) were significantly worse than population norms (P < .05). Fatigue severity was associated significantly (P < .01) with all quality-of-life scores (-0.4 < r < -0.7). In conclusion, severe fatigue and diminished quality of life are more frequent among children with hereditary motor and sensory neuropathy 1A compared to healthy peers. The strong association between fatigue severity and quality of life suggests a negative impact of fatigue on quality of life in these children
AB - Severe fatigue and low quality of life are reported by a majority of adult patients with hereditary motor and sensory neuropathy 1A. In children with hereditary motor and sensory neuropathy 1A, the prevalence and impact of fatigue have not been studied yet. In this questionnaire survey, 55 Dutch children (response rate 77%) with genetically confirmed hereditary motor and sensory neuropathy 1A participated (mean age 15 years [standard deviation 2.1]). Prevalence of severe fatigue (based on a cut-off score of the Checklist Individual Strength) was 24%, in contrast to 14% in a Dutch school-based population (P < .05). Almost all quality-of-life scores (measured with the Child Health Questionnaire-Child Form 87) were significantly worse than population norms (P < .05). Fatigue severity was associated significantly (P < .01) with all quality-of-life scores (-0.4 < r < -0.7). In conclusion, severe fatigue and diminished quality of life are more frequent among children with hereditary motor and sensory neuropathy 1A compared to healthy peers. The strong association between fatigue severity and quality of life suggests a negative impact of fatigue on quality of life in these children
U2 - https://doi.org/10.1177/0883073812447681
DO - https://doi.org/10.1177/0883073812447681
M3 - Article
C2 - 22752492
SN - 0883-0738
VL - 28
SP - 429
EP - 434
JO - Journal of child neurology
JF - Journal of child neurology
IS - 4
ER -