TY - JOUR
T1 - Preliminary development and validation of the Indonesian Pediatric Epilepsy Questionnaire (INA-PEPSI) to determine epilepsy and distinguish focal and generalized epilepsy in infants and children with unprovoked seizure in low-resource settings
AU - Handryastuti, Setyo
AU - Tiansyah, Rizal Agus
AU - Mangunatmadja, Irawan
AU - Saputra, Deddy R.
AU - Octaviana, Fitri
AU - Budikayanti, Astri
AU - Alatas, Fatima Safira
AU - Pusponegoro, Hardiono D.
AU - Tridjaja, Bambang
AU - Soebandi, Amanda
N1 - Publisher Copyright:
© 2024 The Author(s). Epilepsia Open published by Wiley Periodicals LLC on behalf of International League Against Epilepsy.
PY - 2024
Y1 - 2024
N2 - Objective: To outline the preliminary development and validation of a questionnaire for diagnosing epilepsy and distinguishing focal and generalized epilepsy among infants and children in Indonesia, where electroencephalography and pediatric neurologists are generally not available. Methods: A 10-question questionnaire comprising of 43 items was developed through literature review and expert panel discussions. Then, the questionnaire was administered by pediatricians to 75 children aged 1 month to 18 years old presenting with >1 episode of unprovoked seizures at an interval of >24 h. Subsequently, the questionnaire was assessed for content validity with item-level and scale-level content validity indices and ratio, construct validity with item-total correlation tests, criterion validity with diagnostic parameter assessments, and inter-rater reliability using Cohen's kappa (κ) and internal consistency with Cronbach's alpha (α) coefficient. Results: The questionnaire exhibited favorable internal validity and reliability in diagnosing epilepsy and distinguishing focal and generalized epilepsy, with excellent content (both indices and ratio at 1) and construct validity (rcount > rtable at p < 0.001), inter-rater reliability (κ = 0.86 and κ = 0.84), and internal consistency (α = 0.634 and α = 0.806). The questionnaire had a sensitivity and specificity of 96.4% (95%CI 89.1–99.5%) and 95.0% (79.5–99.6%) (area under the curve [AUC] 0.946 [0.900–0.992, p < 0.001]) in diagnosing epilepsy and 80.0% (57.4–95.7%) and 97.4% (89.7–99.2%) (AUC 0.889 [0.783–0.995, p < 0.001]) in distinguishing focal and generalized epilepsy, with a misdiagnosis rate of 4.0%. Significance: The questionnaire shows promising potential in diagnosing epilepsy and distinguishing focal and generalized epilepsy. Further external validation studies in larger and more diverse populations are required to confirm our findings. Plain Language Summary: The diagnosis of epilepsy in children is challenging, particularly in resource-limited settings such as Indonesia, where advanced diagnostic tests and pediatric neurologists are scarce. The Indonesian Pediatric Epilepsy Questionnaire (INA-PEPSI) is designed to address these limitations by enabling healthcare professionals in Indonesia to diagnose epilepsy and classify its types without relying on advanced diagnostic tools. Although the questionnaire is still in the early stages of development and validation, this study demonstrates that the questionnaire exhibits good overall diagnostic performance in diagnosing epilepsy and distinguishing epilepsy types among Indonesian children.
AB - Objective: To outline the preliminary development and validation of a questionnaire for diagnosing epilepsy and distinguishing focal and generalized epilepsy among infants and children in Indonesia, where electroencephalography and pediatric neurologists are generally not available. Methods: A 10-question questionnaire comprising of 43 items was developed through literature review and expert panel discussions. Then, the questionnaire was administered by pediatricians to 75 children aged 1 month to 18 years old presenting with >1 episode of unprovoked seizures at an interval of >24 h. Subsequently, the questionnaire was assessed for content validity with item-level and scale-level content validity indices and ratio, construct validity with item-total correlation tests, criterion validity with diagnostic parameter assessments, and inter-rater reliability using Cohen's kappa (κ) and internal consistency with Cronbach's alpha (α) coefficient. Results: The questionnaire exhibited favorable internal validity and reliability in diagnosing epilepsy and distinguishing focal and generalized epilepsy, with excellent content (both indices and ratio at 1) and construct validity (rcount > rtable at p < 0.001), inter-rater reliability (κ = 0.86 and κ = 0.84), and internal consistency (α = 0.634 and α = 0.806). The questionnaire had a sensitivity and specificity of 96.4% (95%CI 89.1–99.5%) and 95.0% (79.5–99.6%) (area under the curve [AUC] 0.946 [0.900–0.992, p < 0.001]) in diagnosing epilepsy and 80.0% (57.4–95.7%) and 97.4% (89.7–99.2%) (AUC 0.889 [0.783–0.995, p < 0.001]) in distinguishing focal and generalized epilepsy, with a misdiagnosis rate of 4.0%. Significance: The questionnaire shows promising potential in diagnosing epilepsy and distinguishing focal and generalized epilepsy. Further external validation studies in larger and more diverse populations are required to confirm our findings. Plain Language Summary: The diagnosis of epilepsy in children is challenging, particularly in resource-limited settings such as Indonesia, where advanced diagnostic tests and pediatric neurologists are scarce. The Indonesian Pediatric Epilepsy Questionnaire (INA-PEPSI) is designed to address these limitations by enabling healthcare professionals in Indonesia to diagnose epilepsy and classify its types without relying on advanced diagnostic tools. Although the questionnaire is still in the early stages of development and validation, this study demonstrates that the questionnaire exhibits good overall diagnostic performance in diagnosing epilepsy and distinguishing epilepsy types among Indonesian children.
KW - children
KW - diagnosis
KW - epilepsy
KW - questionnaire
KW - seizures
UR - http://www.scopus.com/inward/record.url?scp=85200975088&partnerID=8YFLogxK
U2 - 10.1002/epi4.13021
DO - 10.1002/epi4.13021
M3 - Article
C2 - 39110085
AN - SCOPUS:85200975088
SN - 2470-9239
VL - 9
SP - 1868
EP - 1880
JO - Epilepsia Open
JF - Epilepsia Open
IS - 5
ER -