Validation of the Neuro-QoL measurement system in children with epilepsy
Introduction
The prevalence of epilepsy among patients seen in pediatric neurology practice may be as high as 70% [1], and these children often experience a variety of comorbid conditions such as attention problems and cognitive deficits [2]. Children with epilepsy often face complex psychosocial consequences that can derail the course of normal development, often extending to adulthood [3], [4]. They have approximately a threefold increased risk of subnormal mental ability or other learning and behavior problems [1], [5], [6], [7], twice the referral rate for mental health services, and a threefold increase in utilization of special education services [8], [9]. Adolescents with epilepsy are also noted to have a higher frequency of behavioral problems than peers who are healthy or have other chronic health problems [10]. Thus, it is important to monitor their health-related quality of life (HRQL) regularly.
Several factors complicate the assessment of HRQL in children and adolescents with epilepsy, including development-related change in basal functioning; difficulties associated with proxy assessment; limitations related to learning ability, behavioral disorders, and motor handicap; and the episodic nature of the disease [5]. Recently, pediatric and adolescent measures have been designed to address the above concerns. Carpay et al. [11] developed a seizure severity (SS) and side effects (SE) scale for children, designed for parent completion. The Quality of Life in Childhood Epilepsy (QOLCE) questionnaire by Sabaz et al. [12] is a parent report scale sampling five core functional domains across 16 subscales. Adolescent self-report measures include the Adolescent Psychosocial Seizure Inventory [13], which does not offer a complete HRQL assessment, and the more comprehensive Quality of Life in Epilepsy for Adolescents inventory (QOLIE-AD-48) [12], [14], [15].
From 2004 to 2009, the National Institute of Neurologic Disorders and Stroke (NINDS) sponsored a multisite project to develop a clinically relevant and psychometrically sound HRQL measurement tool for adults and children with neurological disorders [16], [17], [18], [19]. This effort, Neuro-QoL, enables clinical researchers to compare the HRQL impact of different interventions within and across various conditions. The pediatric Neuro-QoL consists of ten self-report measures: 8 item banks (anxiety, depression, anger, interaction with peers, fatigue, pain, cognitive function, and stigma) and 2 scales (upper and lower extremity functions). These measures are applicable for use with children ages 8 through 17 years and available in both English and Spanish. Different from other HRQL measures used with patients with epilepsy, all Neuro-QoL item banks were developed using rigorous qualitative and quantitative approaches and were calibrated using item response theory (IRT) models [20], [21], [22]. An item bank provides the foundation for the development of dynamic computer adaptive testing (CAT), as well as the creation of a variety of static, fixed-length short forms. The scores produced by any of the instruments created from an item bank are calibrated on the same latent trait and are comparable regardless of the specific questions asked of a given individual or group of respondents. Fixed-length short forms, in which a subset of bank items can be selected from across the trait spectrum to produce a static instrument, can be used when access to computers is limited. When computers are available, CAT, a dynamic process of test administration in which items are selected on the basis of patients' responses to previously administered items, can provide brief-yet-precise measures [23], [24], [25], [26]. The purpose of this paper is to report the validation of Neuro-QoL fixed-length forms in a sample of children with epilepsy.
Section snippets
Pediatric Neuro-QoL measurement system
Details of pediatric Neuro-QoL development have been previously described in Lai et al. [17]. In brief, items were generated by gathering concerns from parents, patients, and clinicians. Because generic item sets (pools) could be answered by a person without a medical condition, generic domains were field-tested on samples from the US pediatric general population. Targeted item pools, typically symptoms or side effects of a disease process (i.e., fatigue, cognitive function, stigma, and pain in
Subjects
Participants (N = 61) were primarily male (62.3%), white (75.9%), and non-Hispanic (79.3%) with average age = 13.4 years (SD = 2.6; range = 10 to 18 years). At time 1, 91.8% were a full-time student (1.6% part-time student and 6.6% did not attend school). Average time since diagnosis was 5 years (SD = 4.1, range = 0.2 to 14.8 years). Medical records revealed that most patients (64.2%) had not experienced a seizure in the past 3 months; 17.8% reported having a seizure daily, 13.3% weekly, 35.6% monthly, and
Discussion
Childhood epilepsy may impact several key aspects of patients' well-being such as cognition and physical, emotional, and social functioning. A valid and reliable measure can facilitate understanding of the impact of childhood epilepsy upon QOL and also enable prompt interventions. The Neuro-QoL measurement system is clinically relevant and psychometrically sound, providing a core set of questions sampling domains that are relevant to patients with many chronic neurological diseases. This paper
Conclusion
In conclusion, pediatric Neuro-QoL is a valid and reliable measure of quality of life for children with epilepsy. The validity of the pediatric Neuro-QoL needs to be evaluated further by recruiting a more diverse sample across the severity spectrum. Since the adult version of the Neuro-QoL was validated on patients with epilepsy [19], the same measurement system can be used to monitor these children throughout the lifespan in a consistent manner.
The pediatric Neuro-QoL can be used in research
Acknowledgments
This manuscript was supported the National Institute of Neurological Disorders and Stroke (HHSN271201200036C; PI: David Cella) and National Cancer Institute (R01CA125671; PI: Jin-Shei Lai).
Conflict of interest
All authors report no conflict of interest to be disclosed.
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